REFERENCIAS BIBLIOGRÁFICAS / BIBLIOGRAPHICAL REFERENCES

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1.) Arthropathy associated with cystic acne, hidradenitis suppurativa, and perifolliculitis capitis abscedens et suffodiens: treatment with isotretinoin. 

2.) Psoriatic arthritis and the spectrum of syndromes related to the SAPHO(synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome. 

3.) SAPHO syndrome or psoriatic arthritis? A familial case study. 

4.) SAPHO syndrome of the temporomandibular joint associated with sudden deafness. 

5.) The SAPHO syndrome in children: a rare cause of hyperostosis and osteitis. 

6.) Cervical spinal cord injury in sapho syndrome. 

7.) Thrombosis of the subclavian vein in SAPHO syndrome. A case-report. 

8.)Vertebral involvement in SAPHO syndrome: MRI findings. 

9.) SAPHO syndrome: 20-year follow-up. 

10.) Stress-induced SAPHO syndrome. 

11.) Recurrent subcutaneous abscess of the sternal region in ulcerative colitis. 

12.) [Vertebral destruction with severe pain in the SAPHO syndrome]. 

13.) [The SAPHO syndrome: a study of 16 cases]. 

14.) [Spondarthritis hyperostotic pustulo-psoriatica: nosologic study with clinical and radiologic presentation in relation to the SAPHO syndrome]. 

15.) [The synovitis-acne-pustulosis-hyperostosis-osteitis (SAPHO) syndrome, a rare extra-digestive manifestation of Crohn's disease. Presentation of 1 case and review of the literature]. 

16.) [Inflammatory spondylodiscitis as a unique radiological manifestation of the SAPHO syndrome]. 

17.) The SAPHO syndrome: defining the radiologic spectrum of diseases comprising the syndrome. 

18.) Synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome: a brief review of a rare condition. 

19.) Two cases of discitis due to Propionibacterium acnes. 

20.) Is SAPHO syndrome a spondyloarthropathy? A vasculopathy? Report of a case. 

21.) Chronic recurrent multifocal osteomyelitis associated with Crohn's disease. 

22.) Osteitis--an under-recognised association with seronegative spondyloarthropathy? 

23.) [Garre sclerosing osteomyelitis]. 

24.) Anterior chest wall arthritis and osteitis associated with Sneddon- Wilkinson disease. 

25.) Chronic diffuse sclerosing osteomyelitis of the mandible or mandibular location of S.A.P.H.O. syndrome. 

26.)[SAPHO syndrome: common denominator for various bone and skin diseases] 

27.)[SAPHO syndrome. Case description of 3 patients with acne conglobata and osteoarticular symptoms] 

28.)Case report. synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome. 

29.) [Chronic recurrent multifocal osteomyelitis in children] 

30.) Acquired hyperostosis syndrome--AHYS--(sternocostoclavicular hyperostosis, pustulotic arthro-osteitis, SAPHO-syndrome): bone scintigraphy of the anterior chest wall. 

31.) The course of the SAPHO syndrome during pregnancy. 

32.)[Lyme disease and SAPHO syndrome] 

33.) Spondylodiscitis in SAPHO syndrome. A series of eight cases. 

34.) [Lumbo-sacroiliac hyperostosis with retroperitoneal fibrosis in spondarthritis hyperostotical pustulo-psoriatica--with sternocostoclavicular hyperostosis and superior and inferior venous obstruction syndrome: pathogenetic hypothesis of fibro-osteopathia psoriatica] 

35.) Sapho syndrome associated with acne fulminans and prominent acromioclavicular joint involvement. 

36.) An aseptic inflammation of the clavicle in a patient with Crohn's disease. A potential manifestation of the SAPHO syndrome. 

37.) SAPHO syndrome: a follow up study of 19 cases with special emphasis on enthesis involvement. 

38.) SAPHO syndrome. 

39.) Psoriatic arthritis and synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome. 

40.) The SAPHO syndrome. 

41.) SAPHO syndrome [see comments] 

42.) SAPHO syndrome. 

43.) Is diffuse sclerosing osteomyelitis of the mandible part of the synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome? Analysis of seven cases 

44.) Sternoclavicular pustulotic osteitis (SAPHO). 20-year interval between skin and bone lesions. 

45.) Recent developments in psoriatic arthritis. 

46.) Miscellaneous neurologic, cardiac, pulmonary, and metabolic disorders with rheumatic manifestations. 

47.) [Lympho Plasma Cellular osteomyelitis] 

48.) [Primary chronic osteomyelitis] 

49.) Aseptic spondylitis as the initial manifestation of the SAPHO syndrome. 

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1.) Arthropathy associated with cystic acne, hidradenitis suppurativa, and perifolliculitis capitis abscedens et suffodiens: treatment with isotretinoin. 

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Cutis 1999 Aug;64(2):87-90 

Libow LF, Friar DA 

Armed Forces Institute of Pathology, Dermatopathology Division, Washington, 

DC 20306-6000, USA. 

A patient with arthropathy associated with cystic acne, hidradenitis 

suppurativa, and perifolliculitis capitis abscedens et suffodiens who 

showed a dramatic response to isotretinoin is described. This, to our 

knowledge, is the first report documenting effective treatment of this 

condition, whose nosologic position with respect to other 

spondyloarthropathies associated with cutaneous disease is considered. 

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2.) Psoriatic arthritis and the spectrum of syndromes related to the SAPHO(synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome. 

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Curr Opin Rheumatol 1999 Jul;11(4):251-6 

Winchester R 

Division of Autoimmune and Molecular Diseases, Columbia University College  of Physicians and Surgeons, New York, New York 10032, USA. 

During the past year, the increasing use of nuclear magnetic resonance  imaging techniques, with their ability to delineate cartilage and  ligamentous structures and to identify edema, are providing a radical  improvement in ascertainment of musculoskeletal abnormalities, although  their significance remains incompletely delineated.

A second theme has come  from the study of spondyloarthropathies in different ethnic groups and  societal environments, revealing that the Northern European and North  American form of the disease, with its powerful association with HLA-B27,  is little evident in the rest of the world's population and that different  susceptibility genes and environmental factors operate in other regions and  peoples. Related to this theme is the compelling evidence of the marked  influence of HIV infection on the development of spondyloarthropathies in  Africa. Two areas of immune recognition are discussed as examples of  emerging fields that may provide useful paradigms for the experimental  approach to mechanisms in psoriatic arthritis.

One of these is the  three-cell model of CD8 T-cell interaction, in which a dendritic cell  presents a peptide from an immunogenic protein to both a CD4 and CD8 T-cell  clone, providing a cognitive interaction that disrupts tolerance and  results in the expansion of the cytotoxic T-cell clone. In this respect,  the combination of an activated dendritic cell, together with enhanced  availability of arthritogenic microbial antigens caused by microbial  persistence, are interesting candidates to explore as the basis of the  HIV-associated rheumatic diseases.

The second area of immune recognition is  the growing understanding of the outline of the solution to the problem of  the association of a spondyloarthropathy with several 

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3.) SAPHO syndrome or psoriatic arthritis? A familial case study. 

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Rheumatology (Oxford) 1999 May;38(5):463-7 

Dumolard A, Gaudin P, Juvin R, Bost M, Peoc'h M, Phelip X 

Department of Rheumatology, CHU, Grenoble, France. 

OBJECTIVE: To discuss the relationships between SAPHO (synovitis, acne,  pustulosis, hyperostosis and osteitis) syndrome and the group of  spondyloarthropathies.

METHODS: Few reports of familial SAPHO have been  published. We describe three children, two sisters and one brother, whose  clinical and radiological presentation was in accordance with SAPHO  syndrome.

RESULTS: Two children developed psoriasis, and one child  palmoplantar pustulosis. Both sacroiliac and sternoclavicular joints were  involved in these three cases. Some features in our observations are also  common to psoriatic arthritis. No association was found with HLA antigens,  but a history of trauma preceding the onset of symptoms was present in all  three children.

CONCLUSIONS: We can consider that SAPHO is nosologically  related to spondyloarthropathies. Psoriatic arthritis could be the missing  link between SAPHO and spondyloarthropathies. It is likely that both genetic  and environmental factors are involved. 

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4.) SAPHO syndrome of the temporomandibular joint associated with sudden deafness. 

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AJNR Am J Neuroradiol 1999 May;20(5):902-5 

Marsot-Dupuch K, Doyen JE, Grauer WO, de Givry SC 

Department of Radiology, University Hospital St Antoine, Paris, France. 

We report a case of arthritis of the temporomandibular joint (TMJ)  associated with sclerosing osteomyelitis of the mandible and temporal bone,  causing deafness.

The presence of a palmoplantar pustulosis established the  diagnosis of SAPHO syndrome. SAPHO (an acronym referring to synovitis,  acne, palmoplantar pustulosis, hyperostosis, and osteitis) syndrome is  defined by the association of characteristic osteoarticular and  dermatologic manifestations, with diffuse sclerosing osteomyelitis of the  mandible being a part of this entity. We review the literature of SAPHO  syndrome with mandibular manifestations and discuss the mechanisms of  inflammatory spread from the TMJ to the cochlea.

To our knowledge, this is  the first description of skull base involvement in a patient with SAPHO  syndrome leading to sudden deafness. 

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5.) The SAPHO syndrome in children: a rare cause of hyperostosis and osteitis. 

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J Pediatr Orthop 1999 May-Jun;19(3):297-300 

Letts M, Davidson D, Birdi N, Joseph M 

Department of Pediatrics, Children's Hospital of Eastern Ontario, Ottawa,  Canada. 

The SAPHO syndrome is a rare constellation of signs and symptoms  characterized by synovitis, acne, pustulosis, hyperostosis, and osteitis.  The most common musculoskeletal complaints are hyperostosis, causing pain,  tenderness, and swelling of the anterior chest wall, although any part of  the axial and appendicular skeleton may be affected.

There is a great  degree of variability in the dermatologic involvement of this syndrome. A  combination of clinical, radiographic, and pathological investigation is  required to establish the correct diagnosis. No single treatment has been  found to be effective, although nonsteroidal antiinflammatory drugs have  been the most frequently used.

Because there is no mention of SAPHO  syndrome in the English orthopaedic literature, and pediatric orthopaedic  surgeons may be the first caregivers to treat these children, we thought it  appropriate to share our experience with a 5-year-old boy with SAPHO  syndrome recently under our care. 

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6.) Cervical spinal cord injury in sapho syndrome. 

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Spinal Cord 1999 Apr;37(4):301-4 

Deltombe T, Nisolle JF, Boutsen Y, Gustin T, Gilliard C, Hanson P 

Department of Physical Medicine and Rehabilitation, Mont-Godinne University  Hospital UCL, Yvoir, Belgium. 

Cervical spinal fracture and pseudarthrosis are previously described causes  of spinal cord injury (SCI) in patients with spondylarthropathy. SAPHO  (Synovitis Acne Pustulosis Hyperostosis Osteitis) syndrome is a recently  recognized rheumatic condition characterized by hyperostosis and  arthro-osteitis of the upper anterior chest wall, spinal involvement  similar to spondylarthropathies and skin manifestations including  palmoplantar pustulosis and pustular psoriasis.

We report the first case of  SAPHO syndrome disclosed by SCI related to cervical spine ankylosis. 

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7.) Thrombosis of the subclavian vein in SAPHO syndrome. A case-report. 

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Rev Rhum Engl Ed 1999 Mar;66(3):173-6 

Lazzarin P, Punzi L, Cesaro G, Sfriso P, De Sandre P, Padovani G, Todesco S 

Division of Rheumatology, University of Padova, Italy. 

The hallmark of SAPHO Syndrome (Synovitis, Acne, Pustulosis, Hyperostosis,  Osteitis) is osteitis of the anterior chest wall, which can be so  pronounced as to cause thoracic outlet syndrome, thrombosis of the  subclavian vein, and compression of the superior vena cava. Suggestive skin  manifestations, namely palmoplantar pustulosis and severe acne, generally  antedate the bone and joint lesions.

We report a case of SAPHO syndrome  that went unrecognized for many years and eventually caused compression and  thrombosis of the right subclavian vein requiring cleidectomy. SAPHO  syndrome should be considered in every patients with anterior chest wall  inflammation, particularly when skin lesions are also present. 

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8.)Vertebral involvement in SAPHO syndrome: MRI findings. 

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Skeletal Radiol 1999 Mar;28(3):163-8 

Nachtigal A, Cardinal E, Bureau NJ, Sainte-Marie LG, Milette F 

Department of Radiology, Hopital Saint-Luc, Centre Hospitalier de  l'Universite de Montreal, QC, Canada. 

We report on the MRI findings in the vertebrae and surrounding soft tissues  in two patients with the SAPHO syndrome (Synovitis, Acne, Pustulosis,  Hyperostosis, Osteitis). The MRI findings include abnormal bone marrow  signal, either focal or diffuse, of the vertebral bodies and posterior  elements; hyperintense paravertebral soft tissue swelling and abnormal  signal of the intervertebral discs. These changes are consistent with  discitis and osteitis. 

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9.) SAPHO syndrome: 20-year follow-up. 

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Skeletal Radiol 1999 Mar;28(3):159-62 

Davies AM, Marino AJ, Evans N, Grimer RJ, Deshmukh N, Mangham DC 

Department of Radiology, Royal Orthopaedic Hospital, Birmingham, UK. 

Considerable attention has been paid in the past 10 years to the  radiological spectrum of disease entities belonging to the SAPHO syndrome.  We report an unusual case presenting with an extra-axial (femoral) lesion,  prior to description of this syndrome, which was radiologically and  histologically mistaken for a parosteal osteosarcoma.

Nineteen years later,  a further lesion developed in the scapula together with the typical  sternoclavicular manifestations, at which stage the correct diagnosis of  SAPHO syndrome was established. 

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10.) Stress-induced SAPHO syndrome. 

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J Eur Acad Dermatol Venereol 1999 Jan;12(1):43-6 

Orion E, Brenner S 

Department of Dermatology, Tel Aviv Sourasky Medical Center, Sackler  Faculty of Medicine, Tel Aviv University, Israel. 

We describe the case of a woman with the classic combination of features of  synovitis-acne-pustulosis-hyperostosis-osteitis (SAPHO) syndrome, including  pustulosis palmo-plantaris and anterior chest wall involvement. The varying  symptomology, etiology and pathogenesis of this syndrome and the  contribution of stress are discussed.

The authors ascribe the dearth of  reported cases to lack of awareness and recognition of SAPHO, and not to  the real incidence of the syndrome. 

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11.) Recurrent subcutaneous abscess of the sternal region in ulcerative colitis. 

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Am J Gastroenterol 1999 Mar;94(3):844-5 

Murata I, Satoh K, Yoshikawa I, Masumoto A, Sasaki E, Otsuki M 

Third Department of Internal Medicine, University of Occupational and  Environmental Health, and The Kyoritsu Hospital, Kitakyushu, Japan. 

An 18-yr-old female patient with extensive ulcerative colitis suffered from  several episodes of recurrent aseptic subcutaneous abscesses of the sternal  region with a course paralleling that of her colitis. The abscess seemed to  occur secondarily to osteomyelitis of the sternum, which is a manifestation  of the synovitis, acne, pustulosis, hyperostosis, and osteomyelitis (SAPHO)  syndrome. 

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12.) [Vertebral destruction with sever pain in the SAPHO syndrome]. 

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Dtsch Med Wochenschr 1999 Feb 5;124(5):114-8 

Hagemann D, Pfaffenbach B, Schmid G, Adamek RJ 

Medizinische Klinik I, Ruhr-Universitat, St. Josef-Hospital, Bochum.  Dirk.Hagemann@ruhr-uni-bochum.de 

HISTORY AND ADMISSION FINDINGS: A 57-year-old man had for the past 18  months complained of recurrent, recently worsening, belt-like backache  radiating ventrally.

On admission a skin rash consisting of blister and  pustules was noted on the palms of both hands. He had pain on pressure over  the right upper abdomen, an enlarged prostate and definite pain on  percussing the vertebral column with restricted movement of the thoracic  vertebral column, but no other physical signs.

INVESTIGATIONS: Radiology  revealed clearly increased sclerosis of several thoracic vertebrae with  osteolytic destruction and a paravertebral soft tissue tumor. Search for a  primary tumor was unsuccessful. Bone scintigraphy demonstrated nuclide  enrichment of the thoracic vertebrae and of the sternoclavicular joints  without increase in the LeukoScan. These findings indicated the diagnosis  of SAPHO syndrome (synovitis-acne-pustulosis-hyperostosis-osteomyelitis). 

TREATMENT AND COURSE: Rapid subjective and objective improvement followed  the administration of clindamycin and ibuprofen.

CONCLUSION: In case of  bone pain of uncertain aetiology, especially when associated with skin  rash, the rare SAPHO syndrome should be considered in the differential  diagnosis, avoiding lengthy diagnostic steps and allowing early treatment. 

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13.) [The SAPHO syndrome: a study of 16 cases]. 

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Med Clin (Barc) 1999 Jan 23;112(2):61-3 

Olive A, Perez-Andres R, Rivas A, Holgado S, Casado E, Guma M, Tena X 

Seccion de Reumatologia, Hospital Universitari Germans Trias i Pujol,  Badalona, Barcelona. 

The SAPHO syndrome is characterized by synovitis, acne, palmo-plantar  pustulosis, hyperostosis and osteitis. SAPHO syndrome has been occasionally  described in Spain. We present our experience of 16 cases with the SAPHO  syndrome diagnosed between 1984-1995.

The predominant clinical symptoms  were: anterior thoracic pain (14 cases), sacroiliac pain (7 cases) and  peripheral arthritis (2 cases). Cutaneous involvement was characterized by  palmo-plantar pustulosis (8 cases) acne (3 cases) and psoriasis (2 cases).  The histocompatibility antigen B27 was negative in all cases. A  computerized tomographic study revealed involvement of  sterno-costo-clavicular and manubriosternal joints (14 cases) and  sacroiliitis (8 cases). 

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14.) [Spondarthritis hyperostotica pustulo-psoriatica: nosologic study with  clinical and radiologic presentation in relation to the SAPHO syndrome]. 

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Rofo Fortschr Geb Rontgenstr Neuen Bildgeb Verfahr 1998 Dec;169(6):576-84 

Schilling F, Kessler S 

Radiologische Universitatsklinik Mainz. 

Spondarthritis hyperostotica pustulo-psoriatica (Spond.hyp.pp-Schilling),  corresponding nosologically to pustulotic arthroosteitis, is a  dermato-skeletal "double system" disease of adults.

It consists of the  triad

(a) palmo-plantar pustulosis (Ppp) or, alternatively,  Konigsbeck-Barber-type psoriasis,

(b) sternocostoclavicular hyperostosis  (SCCH), and

(c) truncal-skeletal changes with syndesmophite-like,  hyperostotic and/or parasyndesmophite-like ossifications of layers of the  anterior vertebral ligament taken together in the sense of a desmophytal  hyperostosis.

There is also a potential for sclerosing inflammatory  arthritis of the sacro-iliac joints and "dry" inflammatory arthritis of  peripheral joints. Thus, the pustulo-psoriatic terrain seems to have a  decisive influence on osseous pathology.

A total of 38 cases from a study  during the years 1982 to 1992 is analysed with regard to morphological  characteristics. Rare cases with diaphyseal and pelvic hyperostotic lesions  subsequent to bland sclerosing osteomyelitis constitute an overlapping  region to chronic recurrent multifocal osteomyelitis (CRMO) and illustrate  the relationship between hyperostotic spondarthritis and CRMO. The  syndromes of "acquired hyperostosis" and "SAPHO", the former more  radiologically oriented and the latter more clinically oriented, together  with mainly CRMO and hyperostotic spondarthritis and its forms, constitute  the "Spond.hyp.pp.".

Although hyperostosis is a guidepost for the  radiologist and SAPHO symptoms are one for the clinician, the syndrome does  not represent a diagnosis by itself and requires further differentiation.  In this report the entity "Spond.hyp. pp." is considered and required  contributions from rheumatologically and osteologically oriented radiologist. 

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15.) [The synovitis-acne-pustulosis-hyperostosis-osteitis (SAPHO) syndrome, a rare extra-digestive manifestation of Crohn's disease. Presentation of 1 

case and review of the literature]. 

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Gastroenterol Clin Biol 1998 Feb;22(2):240-3 

Dharancy S, Talbodec N, Asselah T, Hachulla E, Delaporte E, Cortot A,  Colombel JF 

Service des Maladies d l'Appareil Digestif et de la Nutrition, Hopital  Claude-Huriez, CHRU, Lille. 

We report the case of a 33-year-old man with a severe Crohn's disease since  the age of sixteen. He presented with acne and palmoplantar pustulosis  associated with a right knee synovitis. Investigations revealed a major  axial bone condensation. The association  synovitis-acne-pustulosis-hyperostosis-osteitis leaded to the diagnosis of  SAPHO syndrome associated with Crohn's disease. 

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16.) [Inflammatory spondylodiscitis as a unique radiological manifestation of the SAPHO syndrome]. 

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J Radiol 1998 Apr;79(4):337-40 

Cabay JE, Marcelis S, Dondelinger RF 

Departement d'Imagerie Medicale, Domaine Universitaire du Sart-Tilman B 35,  Liege, Belgique. 

We describe two cases of SAPHO with an exclusive spinal involvement.  Diagnosis was established by sterno-clavicular arthralgias, palmar and  plantar pustulosis and by radiological signs of inflammatory  spondylodiscitis and vertebral osteitis. Spondylodiscitis and medullary  edema resolved as shown by MRI after administration of steroids in one case  and methotrexate in the other. 

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17.) The SAPHO syndrome: defining the radiologic spectrum of diseases comprising  the syndrome. 

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Eur Radiol 1998;8(5):800-6 

Sugimoto H, Tamura K, Fujii T 

Department of Radiology, Jichi Medical School, 3311 Minamikawachi-machi,  Kawachi-gun, Tochigi-ken, 329-04, Japan. 

The objective of our study was to clarify the radiologic spectrum of  disease entities belonging to the SAPHO syndrome (SAPHO being an acronym  for synovitis, acne, pustulosis, hyperostosis, and osteitis). A  retrospective analysis of radiologic data was undertaken to determine the  relationship of the osteoarthritic changes seen in palmoplantar pustulosis  (PPP, n = 179), acne (n = 3), psoriasis vulgaris (PsV, n = 355),  generalized pustular psoriasis (GPP, n = 25), and chronic recurrent  multifocal osteomyelitis (CRMO, n = 4).

Osseous changes of PPP, acne, and  CRMO overlap each other; 7 PPP, 2 acne, and 3 CRMO patients manifested  stenocostoclavicular hyperostosis as well as hyperostosis of the spine,  pelvis, and/or femur. These findings were not seen in either PsV or GPP  patients.

Thirteen PsV and 4 GPP patients had peripheral arthritis and/or  symmetrical sacroiliitis, which were not observed in the PPP, acne, and  CRMO patients. The PPP, acne, and CRMO patients may be grouped as belonging  to the single disease entity, namely SAPHO syndrome.

Our findings do not  support the inclusion of PsV and GPP in the spectrum of this syndrome. 

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18.) Synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome: a  brief review of a rare condition. 

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Ann Acad Med Singapore 1998 Jan;27(1):122-4 

Koh ET 

Department of Rheumatology and Immunology, Tan Tock Seng Hospital, Singapore. 

Synovitis, Acne, Pustulosis, Hyperostosis and Osteitis (SAPHO) syndrome is  an osteoarticular-skin syndrome characterised by sterile inflammatory  arthro-osteitis of the anterior chest wall. It is associated with various  skin conditions including palmoplantar pustulosis, severe acnes and  pustular psoriasis.

A link between SAPHO syndrome and seronegative  spondyloarthropathies has been suggested over the last 10 years. SAPHO  syndrome mainly affects young and middle-aged adults. Treatment of SAPHO  syndrome remains empirical as the underlying aetiopathogenesis is unclear. 

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19.) Two cases of discitis due to Propionibacterium acnes. 

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Rev Rhum Engl Ed 1998 Jan;65(1):68-71 

Crouzet J, Claudepierre P, Aribi EH, Malbrunot C, Beraneck L, Juan LH,  Larget-Piet B 

Rheumatology Department, Corbeil Hospital, Corbeil, France. 

We report two cases of discitis due to Propionibacterium acnes and review  previously published cases of bone and joint infections in which this  organism was recovered as a pure culture. P. acnes is an anaerobic organism  usually considered a normal inhabitant of the skin but capable of producing  a variety of infections including discitis, osteitis, arthritis, and chest  wall osteitis.

Most patients were immunocompetent. A few infections  occurred spontaneously, whereas others were secondary to a break in the  skin or to implantation of foreign material into the body for instance  during internal fixation of a fracture or arthroplasty.

Cases of P. acnes  chest wall infection have been reported in patients with palmoplantar  pustulosis or chronic or multifocal osteitis, supporting a role for P.  acnes in SAPHO syndrome. 

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20.) Is SAPHO syndrome a spondylarthropathy? A vasculopathy? Report of a case. 

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Rev Rhum Engl Ed 1997 Jun;64(6):424-7 

Boonen A, Verwilghen J, Dequeker J, Van der Linden S, Westhovens R 

Department of Rheumatology, Maastricht University Hospital, The Netherlands. 

We describe the case of a patient who had SAPHO syndrome with mediastinal  and retroperitoneal fibrosis. After several years, he developed erosive  polyarthritis and systemic vasculopathy. The possible implications of these  associations are discussed. 

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21.) Chronic recurrent multifocal osteomyelitis associated with Crohn's disease. 

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Am J Med Sci 1998 Feb;315(2):133-5 

Bognar M, Blake W, Agudelo C 

Internal Medicine Department, VA Medical Center, Emory University Hospital,  Atlanta, Georgia, USA. 

Chronic recurrent multifocal osteomyelitis (CRMO) was first described by  Giedion in 1972 as a self-limited relapsing noninfectious inflammatory  condition usually affecting children and adolescents. The association of  CRMO with psoriasis, palmoplantar pustulosis, and SAPHO syndrome  (synovitis, acne, pustulosis, hyperostosis, and osteitis) has been  described.

In this case report, we present a 49-year-old man with CRMO  coexisting with Crohn's disease. To our knowledge, this is the first report  of CRMO with Crohn's disease without the presence of pustular skin lesions  or other features of SAPHO syndrome. 

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22.) Osteitis--an under-recognised association with seronegative  spondyloarthropathy? 

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N Z Med J 1997 Dec 12;110(1057):455-9 

Stebbings S, Highton J, Doyle TC, Jeffery AK 

Department of Medicine, Dunedin School of Medicine. 

AIM: To emphasise osteitis as a feature of the spondyloarthritides.

METHOD:  We describe four cases spanning a spectrum of the spondyloarthritides in  which osteitis was a feature.

RESULTS: One patient had psoriatic arthritis  with palmar-plantar pustular psoriasis and extensive osteitis involving the  tibia and fibula. This case provides a link with two cases with SAPHO  syndrome (synovitis, acne, pustulosis hyperostosis, osteitis) who had  palmar-plantar pustulosis and osteitis.

Many now argue that this syndrome  is a form of spondyloarthritis. The fourth case, which was of particular  interest to us, had enteric reactive arthritis and scintigraphic changes  strongly suggesting the presence of osteitis of individual bones in the  wrist.

CONCLUSION: We propose that these four cases demonstrate that  osteitis may be another feature common to the spondyloarthritides and  SAPHO. Awareness of this may facilitate better documentation of this  feature of the disease. 

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23.) [Garre sclerosing osteomyelitis]. 

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Orthopade 1997 Oct;26(10):902-7 

Vienne P, Exner GU 

Orthopadische Universitatsklinik Zurich. 

Osteomyelitis sclerosans Garre is still not completely understood. In spite  of the fact that it is caused by bacterial infection, in most cases no  bacterial growth can be discovered in culture. Furthermore, it remains open  whether the chronic process is maintained by low-grade persistent infection  or is maintained by itself after sterilization of the infection.

Newer  studies that may prove the presence of bacteria, e.g., by PCR, may  elucidate the causes in the future. Sclerosing bone disease, sometimes  multifocal, which present the same clinical, radiographic and histological  features as chronic sclerosing osteomyelitis may be associated with  diseases such as palmoplantar pustulosis, colitis ulcerosa, Crohn's  disease, etc. and are reported as SAPHO (synovitis acne pustulosis  hyperostosis osteitis) syndrome.

In these diseases bacterial growth is  almost always negative. However, temporary improvement under antibiotic  therapy may be observed. A relationship between typical isolated chronic  sclerosing osteomyelitis and SAPHO syndrome may exist, but the diseases  should at present be differentiated.

Neoplasms such as osteoid osteoma,  Ewing sarcoma, or eosinophilic granuloma may simulate primary sclerosing  osteomyelitis, and pathological examination of peripherally taken specimens  may appear to be compatible with the diagnosis of sclerosing osteomyelitis.  It should be appreciated that malignant tumors--even though this is the  exception--can present a chronic, mild picture.

Treatment of osteomyelitis  sclerosans Garre needs be determined on an individual basis. A conservative  approach with antibiotics usually leads only to temporary pain relief. The  biopsy with opening of the medullary canal is sometimes sufficient for  healing, while in extreme situations only segmental or even complete  resection of the diseased bone case result in a permanent cure. 

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24.) Anterior chest wall arthritis and osteitis associated with  Sneddon-Wilkinson disease. 

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Clin Exp Rheumatol 1997 Nov-Dec;15(6):667-9 

Belfiore N, Caporali R, Borroni G, Montecucco C 

Dipartimento di Medicina Interna e Nefrologia, IRCCS Policlinico S. Matteo,  University of Pavia, Italy. 

We describe a 46-year-old man in whom anterior chest wall arthritis and  clavicular osteomyelitis occurred together with sub-corneal pustular  dermatosis (Sneddon-Wilkinson disease). This observation extends the list  of neutrophilic skin lesions that may be involved in the so-called SAPHO  syndrome. 

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25.) Chronic diffuse sclerosing osteomyelitis of the mandible or mandibular  location of S.A.P.H.O. syndrome. 

============================================================ 

Garcia-Marin F; Iriarte-Ortabe JI; Reychler H 

Section of Oral and Maxillofacial Surgery, Hospital Son Dureta, Palma de  Mallorca,  Spain. 

Acta Stomatol Belg (BELGIUM) Jun 1996 93 (2) p65-71 ISSN: 0001-7000 

Language: ENGLISH 

Document Type: JOURNAL ARTICLE; REVIEW; REVIEW, TUTORIAL 

Journal Announcement: 9711 

Subfile: DENTAL 

The chronic diffuse sclerosing osteomyelitis (CDSO) of the mandible has  been  described as an inflammatory disease characterized by recurrent episodes of  intense  pain in the mandible, often accompanied by trismus, paresthesia and  progressive  mandibular deformity.

The etiopathogeny of this entity is not fully known.  The  differential diagnosis must be carried out very carefully, and the  treatment results  are very disappointing. Recently, evidence that suggests that CDSO may be  the  mandibular location of a more diffuse condition, the synovitis, acne,  pustulosis,  hyperostosis, osteitis (SAPHO) syndrome, has been offered.

We describe two  clinical  cases of CDSO of typical evolution which fulfill the criteria for SAPHO  syndrome,  offering us an occasion for a review of the current literature. (31  References) 

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26.)[SAPHO syndrome: common denominator for various bone and skin diseases]  SAPHO-syndroom: gemeenschappelijke noemer voor enkele bot- en huidziekten. 

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Jahangier ZN; Jacobs JW; Bijlsma JW 

Academisch Ziekenhuis, afd. Reumatologie en Klinische Immunologie, Utrecht. 

Ned Tijdschr Geneeskd (NETHERLANDS) Apr 19 1997 141 (16) p764-9 ISSN:  0028-2162 

Language: DUTCH Summary Language: ENGLISH 

Document Type: 

JOURNAL ARTICLE; REVIEW; REVIEW, TUTORIAL English Abstract 

Journal Announcement: 9709 

Subfile: INDEX MEDICUS 

The acronym 'SAPHO' stands for synovitis, acne, pustulosis, hyperostosis  and  osteitis. Three subtypes of SAPHO can be distinguished which have the  following  features in common: sternoclavicular hyperostosis and sterile inflammatory  lesions in  both bone and skin. These subtypes are: sternocostoclavicular  hyperostosis, chronic  recurring multifocal osteomyelitis, and pustular arthro-osteitis.  Hyperostosis and  osteitic lesions may be similar to those seen in malignant bone tumours.  Synovitis  generally does not lead to bone erosions and one-third of the patients  develop  sacroiliitis.

The SAPHO syndrome and seronegative spondyloarthropathy  share some  common features (a higher prevalence of the HLA-B27 antigen, occurrence of  sacroiliitis and a higher incidence of chronic inflammatory bowel disease and  psoriasis). Aetiology and pathogenesis of SAPHO are unknown; prognosis is  good.

The  SAPHO syndrome often runs a protracted course, with intermittent relapses and  remissions without resulting serious disability. Treatment is aimed only at  symptomatic relief and mainly consists of analgetics and nonsteroidal  antiinflammatory drugs. (37 References) 

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27.) [SAPHO syndrome. Case description of 3 patients with acne conglobata and  osteoarticular symptoms] 

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Das SAPHO-Syndrom. Fallbeschreibung von drei Patienten mit Akne  conglobata und  osteoartikularen Symptomen. 

Gutzmer R; Herbst RA; Kapp A; Weiss J 

Dermatologische Klinik und Poliklinik, Medizinische Hochschule Hannover.  Hautarzt (GERMANY) Mar 1997 48 (3) p186-90 ISSN: 0017-8470  Language: GERMAN Summary Language: ENGLISH  Document Type:  JOURNAL ARTICLE English Abstract  Journal Announcement: 9708 

Subfile: INDEX MEDICUS 

We report three new cases of the SAPHO syndrome. This acronyme consists of  synovitis, acne, pustulosis, hyperostosis and osteitis. Symptoms of this  syndrome,  which may not all be present, are pustulotic skin diseases (pustulosis  palmoplantaris  or severe acne) associated with osteoarticular lesions (mainly  sternoclavicular  hyperostosis, spondylarthropathies or chronic recurrent multifocal  osteomyelitis).  The dermatological aspects of this syndrome are discussed in detail. 

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28.)Case report. synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome. 

============================================================ 

Suei Y; Taguchi A; Tanimoto K; Yamada T; Otani K; Fukuda T 

Department of Oral and Maxillofacial Radiology, Hiroshima University,  School of  Dentistry, Japan. 

Dentomaxillofac Radiol (ENGLAND) Nov 1996 25 (5) p287-91 ISSN: 0250-832X 

Language: ENGLISH 

Document Type: JOURNAL ARTICLE 

Journal Announcement: 9708 

Subfile: ; DENTAL 

A case of the SAPHO (synovitis, acne, pustulosis, hyperostosis and  osteitis)  syndrome in a 35-year-old woman is presented. Ignorance of this entity on  the part  of the physicians treating the patient may have contributed to her having  repeated  diagnostic procedures and treatment, some of which may have been  unnecessary.  Dentists are encouraged to suspect the SAPHO syndrome when they encounter a  patient  with mandibular osteomyelitis together with symptoms involving other bones  and skin  lesions such as pustulosis or psoriasis. 

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29.) [Chronic recurrent multifocal osteomyelitis in children]  Osteite chronique multifocale recurrente de l'enfant. 

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Quelquejay C; Job-Deslandre C; Hamidou A; Benosman A; Adamsbaum C 

Service de Radiologie, Hopital Saint-Vincent-de-Paul, Paris. 

J Radiol (FRANCE) Feb 1997 78 (2) p115-21 ISSN: 0221-0363 

Language: FRENCH Summary Language: ENGLISH 

Document Type: 

JOURNAL ARTICLE; REVIEW; REVIEW, TUTORIAL English Abstract 

Journal Announcement: 9707 

Subfile: INDEX MEDICUS 

We have studied retrospectively a series of 10 children presenting with  chronic  multifocal osteomyelitis (8 girls, 2 boys, 7 to 16 years). All patients  had plain  films, bone scintigraphies and histological studies.

Three had CT scan  and/or MRI.  compared with literature data, we observed only one case of palmoplantar  pustulosis  and only 2 cases of lysis of the medial extremity of the clavicle; in  addition, we  report one case of lateral extremity of the clavicle and 2 vertebral  locations.

The  radiological pattern was typical: at the beginning of the disease, plain  films showed  lytic areas which became progressively osteosclerotic with enlargement of  the bone.  In all the cases, bone scintigraphy revealed high uptake areas which were  often  infraclinical.

The diagnosis was delayed from 3 months to 3 years. This  emphasizes  the difficulty of the diagnosis which relies on the association of clinical,  biological and radiological elements. Biopsies are required to rule out an  infectious bacterial osteomyelitis or a tumoral process. The pathogenesis  of OCMR  remains unknown, but the relation with the SAPHO (synovitis, acne,  pustulosis,  hyperostosis, osteitis) syndrome is general accepted because of the similar  features  of the osteitis.

The long term follow up appears to be uncertain 6 of our  patients  are still symptomatic after five years despite anti inflammatory treatment.  (22 References) 

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30.) Acquired hyperostosis syndrome--AHYS--(sternocostoclavicular hyperostosis,  pustulotic arthro-osteitis, SAPHO-syndrome): bone scintigraphy of the  anterior chest wall. 

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Dihlmann W; Dihlmann SW; Hering L 

Roentgen Institute, General Hospital Barmbek, Hamburg, Germany. 

Clin Rheumatol (BELGIUM) Jan 1997 16 (1) p13-24 ISSN: 0770-3198 

Language: ENGLISH 

Document Type: JOURNAL ARTICLE 

Journal Announcement: 9707 

Subfile: INDEX MEDICUS 

The objective of this report is to provide a description of diagnostically  significant scintigraphically recognizable sites and patterns of acquired  hyperostosis syndrome (AHYS) on the anterior chest wall (ACW), which is  involved in  82% of AHYS patients. In 49/90 of our own AHYS patients, planar bone scans  of the  ACW were performed with the gamma camera, applying an average of 650 MBq of  99mTc-  phosphate complexes. In addition, 53 atraumatic patients with  extrathoracic cancer  were available for routine whole-body scintigraphy.

None of these patients  had  increased uptake identifiable as metastasis clinically or by imaging  modalities in  either the ACW or the rest of the skeleton. The scintigraphic involvement  of the  various morphological ACW structures is described in AHYS.

Moreover,  attention is  called to the diagnostic significance of focal hyperactivities at the  anterior end of  the 2nd-8th rib of adults, which are in the 5th place with respect to their  frequency  in AHYS.

The diagnostic significance of sternocostal-joint involvement in  AHYS can  likewise be recognized by bone-scan scintigraphy and will be discussed. Bone  scintigraphy is more sensitive than radiomorphological imaging in AHYS.  This,  however, only applies under three conditions.

1. The increased radiotracer  uptake in  the upper sternocostoclavicular region must be assessed on both the  anterior and the  posterior view of the ACW scan.

2. In addition to the anterior view of the  routine  scintiscan, further anterior scans with reduced scan time of the gamma  camera are  usually necessary. This ensures better visibility of the involvement of  certain  morphological structures that are important for AHYS diagnosis. Moreover, a  statement can be made about the inflammatory ossifying activity/inactivity  of the  AHYS on ACW.

3. Increased radionuclide uptake in the manubrium sterni and  corpus  sterni on the anterior scan should be verified by additional lateral or  oblique scans  of the thorax (sternum). 

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31.) The course of the SAPHO syndrome during pregnancy. 

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Pons JC; Nekhlyudov L; Delfraissy JF 

Service de Gynecologie-Obstetrique I, Hopital Cochin-Port-Royal, Paris,  France. 

Eur J Obstet Gynecol Reprod Biol (IRELAND) Dec 27 1996 70 (2) p215-6  ISSN: 0301-  2115 

Language: ENGLISH 

Document Type: JOURNAL ARTICLE 

Journal Announcement: 9706 

Subfile: INDEX MEDICUS 

In this report, we describe a case of a pregnancy in a 29 year old patient  suffering from a new syndrome, the SAPHO syndrome (synovitis, acne,  pustulosis,  hyperostosis, osteitis). The SAPHO syndrome improved considerably during  the course  of this pregnancy and treatment with indomethacin ceased during gestation. 

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32.) [Lyme disease and SAPHO syndrome]  Enfermedad de Lyme y sindrome SAPHO. 

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de Vega Santos T; Haro Herrero M; Montolio Donate S; Zamarron Moreno A;  Pascual de  Pablo E; Lopez Lopez C 

Servicio de Medicina Interna, Hospital Comarcal Santiago Apostol, Miranda  de Ebro,  Burgos. 

An Med Interna (SPAIN) Jan 1997 14 (1) p28-30 ISSN: 0212-7199 

Language: SPANISH Summary Language: ENGLISH 

Document Type: 

JOURNAL ARTICLE English Abstract 

Journal Announcement: 9706 

Subfile: INDEX MEDICUS 

Joint manifestation in Lyme disease hare shown similarities to those in  reactive  arthritis. SAPHO syndrome can also be included into the group of  seronegative  spondyloarthrophaties.

It is presented a patient with joint, ocular and  serological  features proper of Lyme disease and after ones suggestive of SAPHO  syndrome, who  responds properly to an antibiotic treatment. It is considered the  possibility that  some patients suffering from SAPHO syndrome can also be suffering from Lyme 

disease. 

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33.) Spondylodiscitis in SAPHO syndrome. A series of eight cases. 

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Toussirot E; Dupond JL; Wendling D 

Department of Rheumatology, University hospital J Minioz, Besancon, France. 

Ann Rheum Dis (ENGLAND) Jan 1997 56 (1) p52-8 ISSN: 0003-4967 

Language: ENGLISH 

Document Type: JOURNAL ARTICLE 

Journal Announcement: 9705 

Subfile: INDEX MEDICUS 

OBJECTIVE: To determine the frequency, clinical features, and  radiological and bone  scintigraphic changes of spondylodiscitis in patients with SAPHO  (synovitis, acne,  pustulosis, hyperostosis, and osteitis).

METHODS: The study was  retrospective. Data  from patients with the diagnostic criteria of SAPHO syndrome were analysed  for  clinical features, biological data (HLA B antigen), and pelvic and spine x  rays.  Spine computed tomography (CT) or magnetic resonance imaging (MRI) were  also examined  in some cases.

RESULTS: 25 patients with a diagnosis of SAPHO were seen  since 1985.  Eight had spondylodiscitis. These included five with palmoplantar  pustulosis, one  with pustular psoriasis, one with psoriasis vulgaris, and one with chronic  recurrent  multifocal osteomyelitis without skin disease. The skin lesion occurred  before the  spondylodiscitis in four cases. Chest wall involvement was observed in  four cases  and sacroiliac joint lesions only in the case with osteomyelitis. HLA B27  was always  negative and HLA B8 was found in four cases. Radiological findings  consisted of  erosive or sclerosing remodelling of endplates with a narrowed disc space;  a reduced  height of the vertebral body was also observed in some cases.

These spinal  lesions  occurred in the three vertebral segments. Multiple sites of  spondylodiscitis in the  same patient were common. Bone scan showed mildly increased uptake and CT  and MRI  were useful for detecting signs of infection such as abscess. Enhanced  signals on T2  weighted sequence or after injection of gadolinium were often observed.  Follow up  study of most of these patients suggests that the prognosis of  spondylodiscitis in  the SAPHO syndrome is favourable.

CONCLUSIONS: Despite few description in  the  literature, spondylodiscitis in the SAPHO syndrome is common (32% in this  series).  These radiological findings are similar to the discovertebral changes of  spondylodiscitis in ankylosing spondylitis, thus giving support to the  relations  between SAPHO syndrome and spondylarthropathies. However, this does not  mean that  the pathogenic mechanisms are the same in these two conditions. 

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34.) [Lumbo-sacroiliac hyperostosis with retroperitoneal fibrosis in spondarthritis hyperostotical pustulo-psoriatica--with sternocostoclavicular hyperostosisand superior and inferior venous obstruction syndrome: pathogenetic hypothesisof fibro-osteopathia psoriatica] 

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Die lumbo-sakro-iliakale Hyperostose mit retroperitonealer Fibrose bei  Spondarthritis hyperostotica pustulo-psoriatica--Mit  sterno-kosto-klavikularer  Hyperostose und mit oberem und unterem venosen Obstruktionssyndrom:  Pathogenetische  Hypothese einer Fibro-Osteopathia psoriatica. 

===========================================================

Schilling F; Schweden F 

Freier wissenschaftlicher Mitarbeiter in der Klinik fur Radiologie,  Johannes-  Gutenberg-Universitat Mainz. 

Z Rheumatol (GERMANY) Sep-Oct 1996 55 (5) p331-47 ISSN: 0340-1855 

Language: GERMAN Summary Language: ENGLISH 

Document Type: 

JOURNAL ARTICLE English Abstract 

Journal Announcement: 9705 

Subfile: INDEX MEDICUS 

We report two cases of retroperitoneal fibrosis which were found in 38  patients  with "spondarthritis hyperostotica pustulo-psoriatica" (spond.hyp.p.-p.).  Spond.hyp.p.-p. is associated with sternocosto-clavicular hyperostosis and  resembles  pustulotic arthroosteitis and SAPHO syndrome. However, as dermatologically  and osteo-  histopathologically documented, it is an enthesiopathic and hyperostotic  form of  spondarthritis psoriatica.

Both cases show a lumbosacroiliac hyperostosis  (LSIH)  which is a rare analogue to sternocosto-clavicular hyperostosis (SCCH).  Facultative  paraosseous connective-tissue proliferation results in a new form of  retroperitoneal  fibrosis with sacroiliac origin. Mediastinal and pelvic masses cause  superior and  inferior vena caval obstruction syndromes. Because of proven psoriasis in  pelvic  bone with para-periostal 

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35.) Sapho syndrome associated with acne fulminans and prominent  acromioclavicular joint involvement. 

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AU: Bolukbas-N; Bolukbas-S; Erel-A; Tali-ET 

AD: Department of Physical Medicine and Rehabilitation, Gazi University 

Medical School, Ankara, Turkey. 

SO: Scand-J-Rheumatol. 1996; 25(3): 180-2 

ISSN: 0300-9742 

PY: 1996 

LA: ENGLISH 

CP: NORWAY 

AB: The clinical and radiological findings of a 33 year-old male patient  with acne fulminans and associated SAPHO syndrome are described. The  patient presented with prominent acromioclavicular joint involvement,  unilateral sacroiliitis and subclavian vein compression which are  relatively uncommon features of this syndrome. 

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36.) An aseptic inflammation of the clavicle in a patient with Crohn's  disease. A potential manifestation of the SAPHO syndrome. 

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AU: Kotilainen-PM; Laxen-FO; Manner-IK; Gullichsen-RE; Saario-RM 

AD: Department of Medicine, University of Turku, Finland. 

SO: Scand-J-Rheumatol. 1996; 25(2): 112-4 

ISSN: 0300-9742 

PY: 1996 

LA: ENGLISH 

CP: NORWAY 

AB: A female patient with Crohn's disease developed an septic  osteoarticular involvement of the left clavicle and sterno- and  acromioclavicular regions. Repeated surgical revisions combined with a  broad-spectrum antimicrobial treatment could not prevent the progress of  the disease. However, the patient started to improve after the diagnosis of  the SAPHO (synovitis, acne, pustulosis, hyperostosis and osteomyelitis)  syndrome was made and the dose of her immunosuppressive therapy increased. 

This patient reminds of the existence of extraintestinal aseptic infections  in association with inflammatory bowel disease (IBD). Moreover, it may  provide further evidence on the significant association between SAPHO and 

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37.) SAPHO syndrome: a followup study of 19 cases with special emphasis on  enthesis involvement. 

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AU: Maugars-Y; Berthelot-JM; Ducloux-JM; Prost-A 

AD: Rheumatology Department, Nantes University Hospital CHU, France. 

SO: J-Rheumatol. 1995 Nov; 22(11): 2135-41 

ISSN: 0315-162X 

PY: 1995 

LA: ENGLISH 

CP: CANADA 

AB: OBJECTIVE: To study the natural history, prognosis, and nosology of  synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome.  METHODS: We performed clinical, radiological, and biological reassessment  of 19 cases of SAPHO syndrome (13 with pustulosis palmaris et plantaris, 3  with acne, 3 with no dermatological manifestations) after a mean disease  course of 12.3 yrs (1-35).

RESULTS: Similar involvement was noted in all  patient groups. Despite persistent remittent attacks in 11/19 cases and new  localizations in 10/19, the prognosis is promising and 18/19 patients  considered that improvement has occurred.

Enthesis involvement,  occasionally associated with osteolysis, was in most cases the first event  leading to hyperostosis, but tended to disappear in the spine and  peripheral joints once synostosis was achieved. The behavior of  osteochondral cartilage was similar to that of the enthesis, whereas the  ribs below T6 were spared.

We report the first descriptions of hyperostosis  of the first costotransversal joint, interspinal ossification, erosion of  wrist bones, followed by synostosis, and formation of bony bridges between  ribs.

CONCLUSION: SAPHO syndrome appears to be a relevant disease process  with good prognosis. Although aseptic osteitis is regarded as the hallmark  of this entity, our followup study strongly suggests that the initial  disease targets are enthesis and related tissues.

Forty-three percent of  our patients met criteria of the European Spondyloarthropathy Study Group  for spondyloarthropathy, but only one had the HLA-B27 phenotype. Thus, it  may be considered that SAPHO syndrome is nosologically related to  spondyloarthropathy, especially those types associated with psoriasis, but  probably differs in pathogenesis. 

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38.) SAPHO syndrome. 

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AU: Cotten-A; Flipo-RM; Mentre-A; Delaporte-E; Duquesnoy-B; Chastanet-P 

AD: Department of Radiology, Hospital B, Centre Hospitalier Regional 

Universitaire de Lille, France. 

SO: Radiographics. 1995 Sep; 15(5): 1147-54 

ISSN: 0271-5333 

PY: 1995 

LA: ENGLISH 

CP: UNITED-STATES 

AB: Palmoplantar pustulosis and severe acne are sometimes associated with  peculiar aseptic skeletal conditions, but such skeletal lesions can be  found without skin lesions. The term SAPHO syndrome has been coined for  this cluster of manifestations. (The acronym SAPHO refers to synovitis,  acne, palmoplantar pustulosis, hyperostosis, and osteitis.)

The most common  site of the disease is the upper anterior chest wall, characterized by  predominantly osteosclerotic lesions, hyperostosis, and arthritis of the  adjacent joints.

Osteosclerosis of the vertebral bodies, hyperostosis, and  erosions of the vertebral plates can be encountered. Unilateral  sacroiliitis is frequently observed. Long bone involvement consists of  osteosclerosis or osteolysis with periosteal new bone formation. Peripheral  arthritis can be present but is rarely associated with joint destruction. 

The pathogenesis of this syndrome remains unknown, but a link with  seronegative spondyloarthropathies is probable. Radiologists should be  aware of this unusual syndrome to avoid misdiagnosis (eg, tumor,  infection), unnecessary surgery, and antibiotic therapy. 

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39.) Psoriatic arthritis and synovitis, acne, pustulosis, hyperostosis,  and osteitis syndrome. 

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SO - Curr Opin Rheumatol 1993 Jul;5(4):428-35 

AU - Kahn MF 

PT - JOURNAL ARTICLE; REVIEW (72 references); REVIEW, TUTORIAL 

AB - Psoriatic arthritis is an inflammatory arthropathy associated with  psoriasis, and its clinical presentation varies from case to case. Distal  interphalangeal involvement is characteristic but not seen in all patients.  Enthesopathy, including that of the spine, is common and contributes to the  classification of psoriatic arthritis as a seronegative  spondyloarthropathy.

The etiopathogenesis of psoriatic arthritis is not  well understood, and evolution as measured by follow-up is variable.  Treatment includes nonsteroidal anti-inflammatory drugs and some of the  drugs used in the treatment of rheumatoid arthritis. Sulfasalazine and, in  the more severe cases, cyclosporine, are being studied for efficacy and  tolerance.

Some cases of psoriatic arthritis are associated with an  inflammatory bone disease, frequently seen on the anterior chest wall,  which is part of the newly described SAPHO (synovitis, acne, pustulosis,  hyperostosis, and osteitis) syndrome. SAPHO syndrome is characterized by  this bone involvement, which can affect the spine and peripheral bones.  Various skin conditions are associated with this syndrome, but they do not  necessarily occur in all cases.

Chronic recurrent multifocal osteomyelitis,  which is seen mostly in children, may be a presentation of SAPHO syndrome.  Associations with sacroiliitis, bowel disease, and psoriasis link SAPHO  syndrome with the spondyloarthropathies. 

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40.) The SAPHO syndrome. 

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SO - Acta Derm Venereol 1993 Jun;73(3):208-9 

AU - Vohradnikova O; Zaruba F; Polaskova S; Hoza J; Leiska A 

PT - JOURNAL ARTICLE 

AB - We report two cases of the SAPHO syndrome (synovitis, acne  conglobata, pustulosis palmoplantaris, hyperostosis and osteitis). This  syndrome has been published in the pediatric and rheumatological literature  in recent years. 

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41.) SAPHO syndrome [see comments] 

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CM - Comment in: Dermatology 1993; 186(3):161-2 

SO - Dermatology 1993;186(3):176-80 

AU - Brandsen RE; Dekel S; Yaron M; Caspi D; Ophir J; Brenner S 

PT - JOURNAL ARTICLE 

AB - Four women with pustulosis palmoplantaris (PPP) and associated  sternocostoclavicular hyperostosis are described. The connection between  the two diseases is not coincidental: it is encountered in 9.4% of PPP  patients, and was reported in the French literature as part of the SAPHO  (synovitis acne pustulosis hyperostosis osteomyelitis) syndrome.

This  syndrome is linked to the spondyloarthropathies, having an increased  prevalence of HLA B27 and occurrence of sacroiliitis. None of our patients  had HLA B27, but HLA A26 was found in the 3 patients examined.

No  conclusion can be drawn from this finding, however, since this antigen is  found in 21.7% of Ashkenazi Jews. Recognition of this syndrome by  dermatologists will improve diagnosis and treatment. 

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42.) SAPHO syndrome. 

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SO - Rheum Dis Clin North Am 1992 Feb;18(1):225-46 

AU - Kahn MF; Chamot AM 

PT - JOURNAL ARTICLE; REVIEW (148 references); REVIEW, TUTORIAL 

AB - SAPHO (synovitis, acne, pustulosis, hyperostosis, osteitis) syndrome  put together osteoarticular lesions described separately under numerous  denominations, such as multifocal osteomyelitis, pustulotic arthroosteitis,  acne rheumatism. The association of sterile inflammatory bone lesions and  neutrophilic skin eruptions is the cornerstone of this new syndrome, which  also has links with spondyloarthropathies and plain psoriasis. 

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43.) Is diffuse sclerosing osteomyelitis of the mandible part of the  synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome?  Analysis of seven cases. 

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SO - Oral Surg Oral Med Oral Pathol 1994 Nov;78(5):594-8 

AU - Kahn MF; Hayem F; Hayem G; Grossin M 

PT - JOURNAL ARTICLE 

AB - Diffuse sclerosing osteomyelitis of the mandible has been described  as a localized disease of unknown origin. We offer evidence that mandibular  involvement in the recently described synovitis acne pustulosis  hyperostosis osteitis syndrome exactly fits the accepted description for  diffuse sclerosing osteomyelitis of the mandible.

The clinical, radiologic,  and pathologic findings in seven such cases are presented and discussed. 

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44.) Sternoclavicular pustulotic osteitis (SAPHO). 20-year interval  between skin and bone lesions. 

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SO - J Rheumatol 1991 Jul;18(7):1104-8 

AU - Kahn MF; Bouvier M; Palazzo E; Tebib JG; Colson F 

PT - JOURNAL ARTICLE 

AB - In 2 cases of palmoplantar pustulosis, a 20-year interval was  observed between the skin lesion and the bone involvement. In one case, the  bone lesion came first. In the other a skin lesion was seen for 3 weeks  without subsequent relapse. The 2 cases underline the multifaceted  presentation of the condition for which we have coined the name synovitis  acne pustulosis hyperostosis osteitis (SAPHO) syndrome. 

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45.) Recent developments in psoriatic arthritis. 

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SO - Curr Opin Rheumatol 1994 Jul;6(4):378-84 

AU - Cuellar ML; Silveira LH; Espinoza LR 

PT - JOURNAL ARTICLE; REVIEW (45 references); REVIEW, TUTORIAL 

AB - Psoriatic arthritis affects 5% to 7% of patients with psoriasis.  Genetic, immunologic, and environmental factors play a role in its  pathogenesis.

The role of inflammatory cytokines has been better defined,  and recent immunohistochemical studies of the synovial membranes have shown  important differences and similarities between psoriatic arthritis and  rheumatoid arthritis.

The association of psoriatic arthritis with  infection, particularly HIV, remains an interesting observation. The most  common clinical presentation appears to be peripheral polyarticular, and  extra-articular manifestations including the SAPHO (synovitis, acne,  pustulosis, hypertosis, and osteitis) syndrome are not common. Methotrexate  and sulfasalazine therapy are effective in patients who do not respond to  nonsteroidal anti-inflammatory drugs. 

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46.) Miscellaneous neurologic, cardiac, pulmonary, and metabolic disorders  with rheumatic manifestations. 

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SO - Curr Opin Rheumatol 1993 Jan;5(1):104-10 

AU - Larkin JG 

PT - JOURNAL ARTICLE; REVIEW (54 references); REVIEW, TUTORIAL  AB - Problems both old and new are featured in this year's selection of  rheumatologic aspects of miscellaneous diseases. Paralysis of one or more  limbs can lead to many musculoskeletal complications, and the approach of  Auguste Dejerine-Klumpke in 1918 can be compared with that of the  present-day physician. The reappearance of rheumatic fever continues to  excite interest.

The specificity of the modified Jones criteria has been  questioned, as have the benefits of long-term antibiotic prophylaxis  following an attack of the disease. Meanwhile, metabolic disorders may be  the first diseases to come under novel scrutiny using the techniques of  genetic engineering, with outstanding possibilities for advancing both  understanding and treatment.

Dermatologic diseases other than psoriasis may  be associated with arthropathy. Many of these symptom complexes may be  variants of the recently described SAPHO (synovitis, acne, pustulosis,  hyperostosis, and osteitis) syndrome. 

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47.) [Lymphoplasmacellular osteomyelitis] 

TO: Lymphoplasmazellulare Osteomyelitis. 

============================================================ 

AU: Krauspe-R; Girschick-H; Huppertz-HI 

AD: Orthopadische Klinik, Konig-Ludwig-Haus, Julius-Maximilians-Universitat  Wurzburg. 

SO: Orthopade. 1997 Oct; 26(10): 894-901  ISSN: 0085-4350 

PY: 1997 

LA: GERMAN; NON-ENGLISH 

CP: GERMANY 

AB: Chronic lymphoplasmacellular osteomyelitis may occur in children,  adolescents and adults, but has not been found in newborns or babies either  in our series or in the literature. Symptoms suggesting an acute disease  like fever are uncommon, but a primary chronic course with symptomatic and  asymptomatic periods is typical.

Pain and swelling are the main symptoms;  painless masses are rare. In children and adolescents the clavicle and  metaphyseal regions of long bones are typical sites of chronic abacterial  osteomyelitis. In adults the clavicles or the first two ribs are mainly  affected with synovitis of the adjacent joints, but the long bones are  rarely involved. Laboratory findings are non-specific but important for the  differential diagnosis. The sedimentation rate and c-reactive protein might  be elevated.

The X-ray examination shows osteolytic, sclerotic or mixed  bony changes and, in case of a diaphyseal involvement, onionskinlike  periosteal bone formation may be present, suggesting a malignant process.  In late stages sclerotic bone formations may be seen as a rest. Uni- or  multifocal lesions can be detected by bonescan, as can asymptomatic  lesions.

Magnetic resonance imaging shows gross signal intensity  differences both in the bone and perifocal soft tissue and involvement of  the synovium with gadolinium DPTA enhancement in T1-weighted images. In  early stages of the disease granulocytes, microabscesses and new bone  formations might suggest bacterial osteomyelitis that cannot be  differentiated by histology.

In intermediate phases lymphocytic and  plasma-cellular infiltrates are found, whereas in late phases sclerotic  bone formations and fibrosis of the bone marrow are seen histologically. In  chronic lymphoplasmacellular osteomyelitis, all clinical, radiological and  histological findings, as well as negative bacteriological cultures, are  mandatory and will allow a definitive diagnosis to be made.

The disease may  be uni- or multifocal, and new bone lesions may occur over time, as well as  skin manifestations, which can be found years before or after bone  involvement. The association with dermatological diseases and/or synovitis  led to the acronym SAPHO syndrome (synovitis, acne, pustulosis,  hyperostosis, osteomyelitis).

For the treatment nonsteroidal  anti-inflammatory drugs are effective for pain relief, reduction of  swelling and dysfunction. Antibiotics have been used in several series and  are not effective. Major surgery is not recommended even for recurrences,  and the prognosis for growth and function is excellent in the long term  despite recurrences over several years. 

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48.) [Primary chronic osteomyelitis] 

TO: Primare chronische Osteomyelitis. 

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AU: Jundt-G; Jani-L 

AD: Knochentumor-Referenzzentrum, Instiut fur Pathologie,  Kantonsspital/Universitatskliniken Basel. 

SO: Orthopade. 1997 Oct; 26(10): 889-93  ISSN: 0085-4350 

PY: 1997 

LA: GERMAN; NON-ENGLISH 

CP: GERMANY 

AB: The term primary chronic osteomyelitis covers a heterogeneous spectrum  of clinical features that should be separated for therapeutic reasons.  Unifocal manifestations should be distinguished from multifocal ones. While  bacteria are often found in the first group, bacteriological investigations  are usually negative in the second group.

Additionally, multifocal forms  often recur (chronic multifocal recurrent osteomyelitis), possess a long  clinical course and may be combined with dermatological disorders (SAPHO  syndrome). A biopsy is often necessary for tumor exclusion and obtaining  material for bacteriology. Histology cannot differentiate between the  respective forms of chronic osteomyelitis. 

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49.) Aseptic spondylitis as the initial manifestation of the SAPHO syndrome. 

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AU: Kotilainen-P; Gullichsen-RE; Saario-R; Manner-I; Kotilainen-E 

AD: Department of Medicine, Turku University Central Hospital, Finland. 

SO: Eur-Spine-J. 1997; 6(5): 327-9  ISSN: 0940-6719 

PY: 1997 

LA: ENGLISH 

CP: GERMANY 

AB: We describe the case of a 61-year-old female patient who presented with  spondylitis of the lumbar spine. Although the microbiological cultures of  the bone biopsy specimens obtained during laminotomy remained negative, the  patient was treated with broad-spectrum antimicrobials for 2 months. Eight  months later she started to suffer from pain and tenderness in her sternum  and the medial portion of her left clavicle.

The findings of computed  tomography and gallium-labelled isotope scan were indicative of  sternoclavicular arthritis. Again, all surgically obtained biopsy specimens  yielded negative results in microbiological studies.

The diagnosis of the  SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteomyelitis)  syndrome was then made based on the clinical presentation with recurrent  sterile osteitis in two characteristic locations, and the patient was  started on immunosuppressive therapy.

This case is a reminder that SAPHO  may sometimes occur without any skin manifestations. Since this type of  patient may be admitted to an orthopedic ward, it is important that  orthopedic surgeons are familiar with the syndrome. 

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DATA-MÉDICOS/DERMAGIC-EXPRESS No 2-(76) 19/10/99 DR. JOSÉ LAPENTA R. 

UPDATED 17 JUNE 2025

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Produced by Dr. José Lapenta R. Dermatologist

Venezuela 1.998-2.025