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Melorheostosis of upper limb: A report of four rare cases.

ABSTRACT: Melorheostosis is a rare chronic bone disease, etiology of which remains unclear. It mostly affects lower limbs and clinical features vary in each case. Radiographs show characteristic "candle wax pattern" of hyperostosis. Herein we report four cases of upper limb affection with their symptoms, radiographic findings and treatment. First case presented with gradually progressive swelling in forearm which was initially misdiagnosed as filariasis. Plain radiograph eventually demonstrated melorheostosis of ulna. Second patient had deformity of index finger and pain in hand which was diagnosed on radiograph of hand. Third case presented with progressive flexion deformity of ring and little finger. Fourth patient had ulnar involvement which was an incidental diagnosis. First three patients were managed with bisphosphonates following which there was significant improvement in pain and fourth case was kept on follow-up as he was asymptomatic.

SUBMITTER: Kumar S

PROVIDER: S-EPMC7026615 | biostudies-literature | 2020 Mar-Apr

REPOSITORIES: biostudies-literature

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Melorheostosis of upper limb: A report of four rare cases.

Kumar Sanjeev S Jain Vijay Kumar VK Prabhakar Rahul R

Journal of clinical orthopaedics and trauma 20190611 2

Melorheostosis is a rare chronic bone disease, etiology of which remains unclear. It mostly affects lower limbs and clinical features vary in each case. Radiographs show characteristic "candle wax pattern" of hyperostosis. Herein we report four cases of upper limb affection with their symptoms, radiographic findings and treatment. First case presented with gradually progressive swelling in forearm which was initially misdiagnosed as filariasis. Plain radiograph eventually demonstrated melorheost ...[more]

PMID: 32099304

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Project description:BackgroundGastrointestinal mucormycosis (GIM) is a rare, opportunistic fungal infection with poor prognosis. Clinically, it is difficult to diagnose GIM owing to its nonspecific clinical symptoms and poor suspicion. The estimated incidence of GIM is inaccurate, and most cases are diagnosed accidentally during surgery or upon postmortem examination. GIM usually occurs in patients with immune deficiencies or diabetes. Here, we report two cases of immunocompetent young patients with GIM who had good prognosis after treatment. Compared to other case reports on GIM, our cases had unusual infection sites and no obvious predisposing factors, which make it important to highlight these cases.Case presentationThe first case was that of a 16-year-old immunocompetent boy who was admitted with gastrointestinal bleeding and perforation due to a gastric ulcer. Strategies used to arrest bleeding during emergency gastroscopy were unsuccessful. An adhesive mass was then discovered through laparoscopy. The patient underwent type II gastric resection. Pathological examination of the mass revealed bacterial infection and GIM. The second case was of a 33-year-old immunocompetent woman with a recent history of a lower leg sprain. The patient subsequently became critically ill and required ventilatory support. After hemodynamic stabilization and extubation, she presented with hematemesis due to exfoliation and necrosis of the stomach wall. The patient underwent total gastrectomy plus jejunostomy. The pathology results revealed severe bacterial infection and fungal infection that was confirmed as GIM. The patient fully recovered after receiving anti-infective and antifungal treatments.ConclusionsNeither patient was immunosuppressed, and both patients presented with gastrointestinal bleeding. GIM was confirmed via pathological examination. GIM is not limited to immunocompromised patients, and its diagnosis mainly relies on pathological examination. Early diagnosis, timely surgical treatment, and early administration of systemic drug treatment are fundamental to improving its prognosis.

Dataset Information

Melorheostosis of upper limb: A report of four rare cases.

Publications

Melorheostosis of upper limb: A report of four rare cases.

Similar Datasets

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