Fibrolipomatous hamartoma of median nerve

Jain, TP; Srivastava, Divya; Mittal, Radhey Shyam; Gamanagatti, Shivanand

doi:10.1111/j.1440-1673.2007.01790.x

Cited by 21 publications

(25 citation statements)

References 7 publications

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“…FLHs are three times more common in the upper limb than the lower limb (29). FLH associated with macrodactyly more commonly occur in women and upper extremities compared with men and lower extremities (30). FLH of the median nerve remains a rare tumour in the pediatric (<18 years of age) population.…”

Section: Discussionmentioning

confidence: 99%

“…Nerve territory-oriented macrodactyly is a common concurrent finding, with 20% to 66% of FLH being associated with such macrodactyly (1,30,31). No hereditary or chromosomal abnormality is reported in these cases.…”

Section: Etiologymentioning

confidence: 99%

“…They may be normal or show soft-tissue thickening, which is nondiagnostic (7). They are, however, useful in evaluating patients presenting with macrodactyly for musculoskeletal evaluation (30).…”

Section: Differential Diagnosismentioning

confidence: 99%

“…2012) restricted to the English-language literature (10,17,22,28,30,(31)(32)(33)(34)(35)(36)(37)(38). Among these publications, no reports of bilateral median nerve involvement were identified.…”

Section: Figure 4) Postoperative Examination Of the Hands Following Bmentioning

confidence: 99%

“…The phalanges are most often affected; however, macrodactyly of the metacarpals has also been reported in an older patient with severe disease (26). When associated with macrodactyly, these neoplasms are most commonly identified in the upper extremities of females (30). Macrodactyly may affect both the radial and ulnar sides of the digit equally or asymmetrically, and is unilateral in 90% of cases (26).…”

Section: Clinical Presentationmentioning

confidence: 99%

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Fibrolipomatous hamartoma of the median nerve: A cause of acute bilateral carpal tunnel syndrome in a three-year-old child: A case report and comprehensive literature review

Senger¹,

Classen²,

Bruce³

et al. 2014

CAN J PLAST SURG

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A lthough carpal tunnel syndrome (CTS) is the most common entrapment neuropathy encountered in an adult surgical practice, it is relatively rare in children. In the majority of adults, CTS is believed to be idiopathic in origin. However, in children, CTS is usually secondary to an underlying pathology, such as lysosomal storage diseases, particularly mucopolysaccharidosis and mucolipidosis. Other causes of CTS in children include genetic bony dysplasia, malformations of the anatomical structures of the carpal tunnel, or enlargement of the median nerve by intraneural lipoma, ganglionic cysts, traumatic neuromas, vascular malformations or neurofibromas. An unusual cause of childhood CTS is fibrolipomatous hamartoma (FLH), a rare benign lesion characterized histologically by the presence of mature fat cells admixed with fibroblastic proliferation within the distorted, expanded nerve bundles.We report the first case in the published literature of acute bilateral CTS secondary to bilateral FLH of the median nerves without macrodactyly in a three-year-old boy who presented with incessant crying due to acute bilateral compression neuropathy. Case presentationA three-year-old boy was brought to the emergency room by his mother due to 'incessant crying' that began earlier that morning. His distress had been progressive since he awoke and, on arrival to the emergency room, he was inconsolable. The mother was unable to identify a precipitating cause. On further questioning, the mother recalled his complaining of discomfort in the right wrist for the past three months. The boy was otherwise healthy, took no medications and his immunizations were up to date.On physical examination, a 4 cm to 5 cm swelling of the flexor aspect of the right wrist and thenar atrophy of this hand were recognized with associated weakness in the hand ( Figures 1A and 1B). Tinel's test was positive. The boy's distress escalated with active and passive movement of the wrist, which impeded the assessment of sensation and range of motion. His physical examination was otherwise unremarkable except for an incomplete syndactyly of the second and third toes.Suspecting a neurofibroma secondary to neurofibromatosis, dermatology and ophthalmology were consulted. A complete workup for the same showed no stigmata of neurofibromatosis. Plastic surgery was subsequently consulted to perform an examination under anesthetic for exploration of the right wrist. Intraoperatively, a fusiform enlargement of the median nerve causing carpal tunnel compression was identified (Figures 2A to 2C). The surrounding tissue was otherwise normal. A biopsy of the median nerve was taken for diagnostic purposes, and a carpal tunnel release (CTR) was performed to alleviate compression symptoms. A three-year-old boy was investigated for inexplicable incessant crying. On examination, his left wrist was mildly swollen (three to four months) and sensitive. Exploration and carpal tunnel decompression of the left wrist with incisional biopsy was performed for the presence of a fusiform swe...

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Section: Discussionmentioning

confidence: 99%

Section: Etiologymentioning

confidence: 99%

Section: Differential Diagnosismentioning

confidence: 99%