Transition from diffuse cellular infiltration to extensive nodular granuloma as a manifestation of isolated sarcoidosis in the hand: A case report

doi:10.1053/jhsu.2003.50077

The Journal of Hand Surgery

Volume 28, Issue 3, May 2003, Pages 439-442

https://doi.org/10.1053/jhsu.2003.50077 Get rights and content

Abstract

A patient with soft-tissue sarcoidosis of the hand is described. She initially presented with diffuse soft-tissue infiltration in the digits. After a 12-year time period without steroid treatment the patient developed extensive, tumor-like nodular masses involving multiple digits with lesions extending to the fingertips. Variable clinical features of the soft-tissue sarcoidosis of the hand have been reported as distinctive, independent manifestations of the disease. A transition from the diffuse cellular infiltration to the nodular, tumor-like lesions in the present case shows that the isolated soft-tissue sarcoidosis of the hand may exhibit variable clinical features in a single case. (J Hand Surg 2003;28A:439-442. Copyright © 2003 by the American Society for Surgery of the Hand.)

Section snippets

Case report

A 54-year-old woman presented in 1990 with complaints of stiffness and painless swelling in her index and middle fingers of both hands. The symptoms had been resistant to anti-inflammatory medication although she was otherwise healthy. Physical examination at the time of initial presentation was normal except for diffuse swelling around the extensor tendons of the index and middle fingers (Figs. 1A, 1B).

. (A, B) Diffuse swelling around the index and long proximal phalanges in 1990. (C) Dorsal

Discussion

Although sarcoidosis commonly presents with the characteristic signs and symptoms, unusual manifestations in the hand without typical pulmonary and hilar lymph adenopathy should not preclude its diagnosis. Isolated sarcoidosis in the hand is rare but the diagnosis should be considered when presented with atypical soft-tissue swelling in the hands. The diagnosis of sarcoidosis should be based on multiple aspects of the diseases, that is, on the radiographic, laboratory, and, more importantly, on

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Cited by (8)

Isolated sarcoid flexor or extensor tenosynovitis of the hand, wrist and distal forearm
2021, Hand Surgery and Rehabilitation
Citation Excerpt :
Tenosynovitis is a common hand presentation usually caused by infection, trauma or rheumatic disorder [1]. Sarcoidosis is a systemic non-caseating epithelioid granulomatous disease of unknown etiology [2] that mostly affects the lungs (90% of cases) [3] and rarely the hands [4]. Sarcoidosis of the hand can involve the skin [5], subcutaneous soft tissue [6] or bone [7] in association with tenosynovitis [8].
Sarcoid tenosynovitis is a rare etiology of chronic tenosynovitis of the hand. Its diagnosis is particularly challenging and must be confirmed by histopathological finding of multiple non-caseating epithelioid granulomatous lesions and giant cells. In this study, we retrospectively investigated cases with isolated sarcoid tenosynovitis of the hand undergoing only tenosynovectomy, and assessed the outcomes. Six patients were included in this study. They were referred to the clinic with painful cystic swelling of the hand/wrist/distal forearm, which was tender but not warm or red, with a continuous burning sensation, numbness and limitation of movement. Preoperative laboratory test results were normal. Ultrasound (US) showed thickened flexor or extensor tendon sheaths with multiple pockets filled with a turbid fluid. Tenosynovectomy was performed as completely as possible, sparing intact tendons, and accumulated fluids were aspirated. Outcome was assessed in terms of sensitivity, range of motion (ROM), pain on visual analogue scale (VAS), Quick Disabilities of the Arm, Shoulder, and Hand (QuickDASH) score, and recurrence rate. At a mean follow-up period of 16.7 months, normal sensation and full ROM were restored in all patients. There was a statistically significant difference between preoperative and postoperative pain VAS and QuickDASH scores. Only two cases had recurrence, with mild pain and positive US findings; both responded well to physiotherapy and medical treatment. Isolated sarcoid tenosynovitis should be considered in the etiological diagnosis of chronic tenosynovitis of the hand. Tenosynovectomy can constitute a single treatment line.
IV, therapeutic study.
La ténosynovite sarcoïdosique est une étiologie rare des ténosynovites chroniques de la main. Son diagnostic est particulièrement difficile et doit être confirmé par la découverte histopathologique de multiples lésions granulomateuses épithélioïdes non caséeuses et de cellules géantes. Dans cette étude, nous avons étudié rétrospectivement des cas de ténosynovite sarcoïdosique isolée de la main n'ayant subi qu'une ténosynovectomie et évalué les résultats. Six patients ont été inclus dans cette étude. Ils avaient été envoyés à la clinique pour une tuméfaction kystique douloureuse de la main/du poignet/de la partie distale de l'avant-bras, qui était sensible mais ni chaude ni rouge, avec une sensation de brûlure continue, un engourdissement et une limitation des mouvements. Les examens biologiques préopératoires étaient normaux. L'échographie (U/S) montrait des gaines des tendons fléchisseurs ou extenseurs épaissies avec de multiples poches remplies d'un liquide trouble. Une ténosynovectomie a été réalisée aussi complète que possible avec préservation des tendons intacts, et les fluides accumulés ont été aspirés. Les résultats ont été évalués en termes de sensation, d'amplitude de mouvement, de douleur à l'aide d'une échelle visuelle analogique (EVA), du score Quick Disabilities of the Arm, Shoulder, and Hand (QuickDASH) et du taux de récidive. Après une période de suivi moyenne de 16,7 mois, une sensibilité normale et une amplitude de mouvement complète étaient restaurées chez tous les patients. Il y avait une différence statistiquement significative entre l’EVA et le score QuickDASH préopératoires et postopératoires. Seuls deux cas ont présenté une récidive avec une douleur légère et des signes échographiques. Les deux ont bien répondu à la kinésithérapie et au traitement médical. La ténosynovite sarcoïdosique isolée doit être envisagée dans le diagnostic étiologique des ténosynovites chroniques de la main. La ténosynovectomie peut être considérée comme une seule ligne de traitement.
Sarcoid flexor tenosynovitis as a single early manifestation of the disease
2012, Journal of Plastic, Reconstructive and Aesthetic Surgery
Citation Excerpt :
Very few cases have been reported in which flexor tenosynovitis was the first presentation of sarcoidosis,1,3,5–9 none having flexor tenosynovitis as the first and only symptom of the disease. Imai et al.3 presented a case of sarcoid tenosynovitis of two extensor tendons, as the first manifestation of sarcoidosis. Their patient also presented bilateral adenopathy on chest radiographs.
Multiple unusual complications after extensive chronic sarcoid tenosynovitis of the hand: A case report
2005, Journal of Hand Surgery
Sarcoidosis is an idiopathic disease characterized by multiple noncaseating granulomatous nodules. Involvement of the hand is extremely uncommon and tenosynovitis of the hand is rare. We present a patient who developed sarcoid tenosynovitis of multiple digits involving both the flexor and extensor tendons. He also developed flexor tendon rupture, subchondral bone erosions, and multiple joint subluxations.
A Single Nail Dystrophy as a Unique Manifestation of Sarcoidosis: A Case Report
2022, Skin Appendage Disorders
Rapid development of sarcoid tenosynovitis
2011, Journal of Clinical Rheumatology
Unusual subcutaneous swellings on the hand as primary presenting feature of sarcoidosis
2011, Journal of Rheumatology

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^☆: No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article.

^☆☆: Supported by grants from the Japan Society for Promotion of Science, a Grant-in-Aid from the Japan Ministry of Education, Uehama Memorial Foundation, and Japan Orthopaedics and Traumatology Foundation.

^*: Reprint requests: Shinji Imai, MD, PhD, Department of Orthopeadic Surgery, Shiga University of Medical Science, Setatsukinowa-cho, Otsu-shi, Shiga-ken, 520-2192, Japan.

^**: 0363-5023/03/28A03-0012$30.00/0

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Original Communications: WristTransition from diffuse cellular infiltration to extensive nodular granuloma as a manifestation of isolated sarcoidosis in the hand: A case report☆,☆☆,*,**

Abstract

Section snippets

Case report

Discussion

Clin Dermatol

J Hand Surg

J Hand Surg

J Hand Surg

J Hand Surg

Ann Hand Surg

J Hand Surg

Original Communications: Wrist
Transition from diffuse cellular infiltration to extensive nodular granuloma as a manifestation of isolated sarcoidosis in the hand: A case report☆,☆☆,*,**