Fibrolipomatous hamartoma in the median nerve in the arm - an unusual location but with MR imaging characteristics: a case report

Nilssøn, Jens; Sandberg, Kristina; Dahlin, Lars B.; Vendel, Nina; Balslev, Eva; Larsen, Lis Olesen; Nielsen, Nikoline Juul

doi:10.1186/1749-7221-5-1

Cited by 14 publications

(12 citation statements)

References 21 publications

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“…The cause of these transformations, however, remains elusive. Some believe this neoplasm is congenital/developmental in origin, whereas others maintain it is an acquired/reactive process due to factors such as nerve irritation or trauma (1,4,5,10,24,40). Those who support the congenital theory, especially in relation to FLH of the median nerve in the carpal tunnel, propose that this patient population is born with an abnormally developed flexor retinaculum or an abnormally developed transverse carpal ligament (26,28,41).…”

Section: Etiologymentioning

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). FLH most often occurs unilaterally (18).…”

Section: Clinical Presentationmentioning

confidence: 99%

“…FLH most often occurs unilaterally (18). These lesions become symptomatic once their size exceeds the passageways through which it passes (40). Because the median nerve is commonly affected, the mass is most often on the volar aspect of the wrist or distal forearm (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: Etiologymentioning

confidence: 99%

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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“…Also there was increased thickness of soft tissues in thenar eminence, mid palm and in thumb with suggestion of hypertrophied subcutaneous fat and fatty infiltration of muscles (Figures 5(c)-(f)) and regional osseous gigantism thumb with premature osteoarthrosis of interphalyngeal joints.Usually patients present with longstanding painless mass. Compression of the affected nerve with paresthesia, motor deficit and pain are known late symptoms[11] [13]. But compressive symptoms may occur only in median nerve involve-M. Aruna Devi, A. Meena DOI: 10.4236/ojim.2018.84022 240 Open Journal of Internal Medicine ment due to encroachment by the flexor retinaculum [11].…”

mentioning

confidence: 99%

Role of Ultrasonography in Evaluation of Peripheral Nerves

Devi¹,

Meena²

2018

OJIM

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Ultrasonography is first line, cost-effective modality in imaging of peripheral nerves. High frequency probes allow high resolution imaging of peripheral nerves located at relatively superficial location. USG can diagnose and localize various pathologies that involve the peripheral nerves such as traumatic, inflammatory, infective, neoplastic, and traumatic neuropathies. USG exceptionally has an important role to play in evaluation of patients with suspected nerve injury, as MRI may not differentiate neural contusion from nerve disruption. Neurapraxic injury is seen as swollen nerve with hypoechoic appearance.

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“…FLH is most commonly seen in the median nerve and rarely in radial, ulnar, and sciatic nerves, nerves of the foot, and cranial nerves [1‐3]. Clinically, FLH may be associated with entrapment findings pertaining to the nerve being involved, mostly in the form of carpal tunnel syndrome.…”

mentioning

confidence: 99%

Fibrolipomatous Hamartoma of the Median Nerve: Comparison of Magnetic Resonance Imaging and Ultrasound

Kara

Özçakar

Ekiz

et al. 2013

PM&R

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Fibrolipomatous hamartoma in the median nerve in the arm - an unusual location but with MR imaging characteristics: a case report

Cited by 14 publications

References 21 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

Role of Ultrasonography in Evaluation of Peripheral Nerves

Fibrolipomatous Hamartoma of the Median Nerve: Comparison of Magnetic Resonance Imaging and Ultrasound

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