Multiple neural fibrolipomas with macrodactyly

Gupta, A. K.; Vasudevan, Geetha; Monappa, Vidya; Bhat, Sudha

doi:10.4103/0019-5154.91851

Cited by 18 publications

(25 citation statements)

References 6 publications

(8 reference statements)

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“…Other authors maintain vehemently that complete excision of this fibrofatty growth is contraindicated because it may cause intolerable sensory or motor disturbances and, therefore, should never be offered as a treatment option for FLH (18,26,43). However, others have suggested that it may be indicated in children younger than two years of age due to their ability to reeducate sensibility (24).…”

Section: Treatmentmentioning

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: Treatmentmentioning

confidence: 99%

“…Pain, tenderness, decreased sensation, paresthesia and/or mass effect are common presenting complaints (3,4,43). Signs and symptoms are usually longstanding, often existing for years before seeking medical attention for a painless mass (2,29,40).…”

Section: Clinical Presentationmentioning

confidence: 99%

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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“…The curiosity regarding the etiology, as well as proper treatment aspect is always there in the mind of clinician; hence discussions are always welcome in such disorders. .Though literature based on certain authors suggest that 85% cases have involvement of median nerve and its digital branches in hand, wrist and forearm [9] but if we glance through the above mentioned few case where 11 cases show lower limb and 5 cases show upper limb involvement by the disorder, it becomes clear that lower limb is commonly involved. Radiological findings do play a major role in correct diagnosis where C.T scan confirms the exact nature of the swelling.…”

Section: Discussionmentioning

confidence: 99%

Macrodystrophia Lipomatosa: A Case Report With Review of Literature

Choudhary¹,

Ray²,

Prabhakaran³

2013

jemds

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“…The median nerve is most commonly involved, but involvement of other nerves has been reported. It is rarely found in the digital nerves at the peripheral level [6][7][8].…”

Section: Discussionmentioning

confidence: 99%

“…Gupta A and others have reported a case of multiple neural fibrolipomas in a child with macrodactyly of both hands [7].…”

Section: Discussionmentioning

confidence: 99%

Neural lipoma in little finger with macrodactyly

Mago¹,

Kochhar²

2017

Surg Rehabil

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Neural lipoma is a rare benign tumour commonly involving the median nerve and other less frequently involved nerves include the ulnar, radial, brachial plexus, superficial peroneal nerve, inferior calcaneal nerve and median plantar nerve. This report describes the rare occurrence of neural lipoma in the digital nerve of little finger associated with digital gigantism of right little finger and ulnar border right hand with no associated motor or sensory deficits.

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Multiple neural fibrolipomas with macrodactyly

Cited by 18 publications

References 6 publications

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

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

Macrodystrophia Lipomatosa: A Case Report With Review of Literature

Neural lipoma in little finger with macrodactyly

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