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Table of Contents
CASE REPORT
Year : 2019  |  Volume : 27  |  Issue : 4  |  Page : 214-216

Lipofibromatous hamartoma in median nerve: A case report and review of the literature


Department of Plastic and Reconstructive Surgery, Tehran University of Medical Sciences, Imam Khomeini Hospital Complex, Tehran, Iran

Date of Submission07-Feb-2019
Date of Acceptance17-Feb-2019
Date of Web Publication26-Sep-2019

Correspondence Address:
Dr. Omid Etemad
Department of Plastic and Reconstructive Surgery, Tehran University of Medical Sciences, Imam Khomeini Hospital Complex, Tehran
Iran
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/tjps.tjps_16_19

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  Abstract 


Lipofibromatous hamartoma is a rare fibro-fatty tumor. This capsulated tumor contains no neural elements. Lipofibroma acts like a benign tumor and may not show any activity for many years. Until 2010, only 88 cases are reported in the literatures. Here, we reported a 33-year-old male with a 10 mm × 30 mm × 40 mm lipofibromatous hamartoma in his left palm. All symptoms disappeared after the surgery.

Keywords: Benign tumor, lipofibromatous hamartoma, median nerve, palm


How to cite this article:
Molaei H, Etemad O, Yavari A. Lipofibromatous hamartoma in median nerve: A case report and review of the literature. Turk J Plast Surg 2019;27:214-6

How to cite this URL:
Molaei H, Etemad O, Yavari A. Lipofibromatous hamartoma in median nerve: A case report and review of the literature. Turk J Plast Surg [serial online] 2019 [cited 2019 Nov 12];27:214-6. Available from: http://www.turkjplastsurg.org/text.asp?2019/27/4/214/267927




  Introduction Top


Hamartoma derived from a Greek word means “failure” is a benign growing tumor which may cause compression in adjacent vessels and nerves.[1] Fibrolipomatous hamartomas (FLH) are a type of nerve hamartomas which most often arise in the median nerve.[2] It needs to be decomposed to prevent nerve damage.[3] Pain, numbness, paresthesia, and carpal tunnel syndrome are commonly side effects of median nerve involvement.[4] There are some advises to treat or not the FLH;[5],[6],[7] which may minimize or eliminate the symptoms of it. In this case report, we present a male case with FLH in the left-hand palm who underwent the nerve decomposition and ligament carpal tunnel release (CTR) surgery. A biopsy was performed for the patient, and he was treated successfully.


  Case Report Top


This case was a 34-year-old male with the 15-year history of a mass in wrist and proximal part of the palm with the progressive course. He experienced occasional pain in hand and fingers which intensified by touching. Physical examinations showed an undefined margin mass in the proximal part of palm with a positive Tinel's Test and no functional impairment. Diagnostic sonography and magnetic resonance imaging (MRI) were performed for this patient [Figure 1]. Sonography reported a mass with dimension of 10 mm × 30 mm × 40 mm in the left palm around the flexor tendons. MRI reported a mass with the dimension of 24 mm × 15 mm at volar side of the left-hand superficial to flexor tendons at the carpometacarpal joint level. According to the findings, the patient was a candidate for the surgery. [Figure 2] shows an image of the patient's hand before the surgery.
Figure 1: Magnetic resonance imaging performance results

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Figure 2: An image of patient's left hand

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The mass position was marked, and the patient underwent the exploration surgery. As shown in [Figure 3]a and [Figure 3]b, a generalized hypertrophy in the median nerve was observed. With the primary diagnosis of lipofibromatous hamartoma, CTR surgery was performed for the patient [Figure 4]a and [Figure 4]b and the biopsy of nerve sheath was taken for more evaluations. He reported no problem in the postoperative progress and all symptoms disappeared after the surgery.
Figure 3: (a) Lipofibromatous hamartia, (b) lipofibromatous hamartoma

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Figure 4: (a and b) Carpal canal release surgery

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Pathological findings reported fibroadipose tissue with dense sclerosis within the median nerve sheath of palm which was in favor of our diagnosis.


  Discussion Top


Lipofibromatous hamartoma is a rare growing fibro-fatty tumor which most commonly affects on the median nerve.[8] LFH presents by pain, paresthesia, and carpal tunnel involvement and can be diagnosed using ultrasonography, computed tomography, and MRI.[7],[8],[9],[10] Radiologists recommend sonography as a diagnostic modality as it is faster, cheaper, and easier to perform in comparison with MRI and may obviate the need for MRI and biopsy.[11] However, at the same time, it is easy to diagnose LFH by MRI due to its serpiginous and hypointense nerve fibers that are interwoven with abundant T1/T2 hyperintense fatty tissue along the median nerve.[4]

These types of tumors do not involve surrounding tissues.[12] The cause of growing this abnormally is still unknown,[13] and there are different theories about it; but in the case of median nerve FLH in carpal tunnel, scientists propose that the patient is born with an abnormally developed flexor retinaculum or transverse carpal ligament.[14],[15],[16]

Fibrolipomatous was first introduced by Mason in 20 century.[17] FLH has been described in infants, children, and adults [15] and in the last case; it may be exist for many years before the symptoms appear.

There are different suggestions for the treatment of FLH such as CTR,[2],[18] total nerve resection in symptomatic patients,[2] and excision of involved nerve segment with or without grafting.[19],[20],[21]

CTR is a most commonly recommended technique in which during a microsurgery, the bulk of the tumor may be reduced.[12],[19],[22],[23],[24] If the bulk of the tumor is small enough, it can be removed without damage to the nerve.[25] Satisfactory results have been achieved by this method in eliminating the symptoms of FLH.[2],[18],[26]

For cases with worsening symptoms, intraneural dissection, nerve resection with grafting and debulking may be helpful.[2],[27],[28] However, this method may lead to tremendous loss of neurological function.[6],[19],[20],[29]

In our case, physical examinations were performed before the surgery. Using ultrasonography and MRI, we diagnosed the existence of FLH in the left-hand palm and the patient candidate for the surgery. Due to the involvement of median nerve and its severe compression under the flexor retinaculum, the carpal tunnel was released by cutting through the ligament that is pressing down on it to make more room for the median nerve and a tendon passing through the tunnel which may lead to reduce the symptoms of disease such as pain and improve the function.

Pathological results of biopsy showed fibroadipose tissue within the median nerve sheath. Excessive proliferation of fibroadipos tissue is a source of LFH in which the epineural and perineural elements of peripheral nerves will be infiltrated.[9],[15],[30] These observations were in favor of our diagnosis.


  Conclusion Top


CTR surgery may be a helpful method in the treatment of lipofibromatous hamartoma and leads to eliminating the symptoms of disease with no damage to the median nerve.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

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Senger JL, Kanthan R, Hamartomas N. Nerve Hamartomas - Fact or Fiction?. Surg Oncol Clin Pract J 2016;1:1-2.  Back to cited text no. 1
    
2.
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3.
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4.
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Patil VS, Nagle S. Lipofibromatous hamartoma of the median nerve. Indian J Plast Surg 2009;42:122-5.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Louis DS, Hankin FM, Greene TL, Dick HM. Lipofibromas of the median nerve: Long-term follow-up of four cases. J Hand Surg Am 1985;10:403-8.  Back to cited text no. 6
    
7.
Clavijo-Alvarez JA, Price M, Stofman GM. Preserved neurologic function following intraneural fascicular dissection and nerve graft for digital and median nerve lipofibromatous hamartoma. Plast Reconstr Surg 2010;125:120e-2e.  Back to cited text no. 7
    
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Wong BZ, Amrami KK, Wenger DE, Dyck PJ, Scheithauer BW, Spinner RJ, Lipomatosis of the sciatic nerve: typical and atypical MRI features. Skeletal Radiol 2006;35:180-4.  Back to cited text no. 8
    
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Al-Jabri T, Garg S, Mani GV. Lipofibromatous hamartoma of the median nerve. J Orthop Surg Res 2010;5:71.  Back to cited text no. 9
    
10.
Toms AP, Anastakis D, Bleakney RR, Marshall TJ. Lipofibromatous hamartoma of the upper extremity: A review of the radiologic findings for 15 patients. AJR Am J Roentgenol 2006;186:805-11.  Back to cited text no. 10
    
11.
Arora R, Arora AJ. Imaging features on sonography and MRI in a case of lipofibromatous hamartoma of the median nerve. Quant Imaging Med Surg 2014;4:207-9.  Back to cited text no. 11
    
12.
Patel ME, Silver JW, Lipton DE, Pearlman HS. Lipofibroma of the median nerve in the palm and digits of the hand. J Bone Joint Surg Am 1979;61:393-7.  Back to cited text no. 12
    
13.
Afshar A. Carpal tunnel syndrome due to lipofibromatous hamartoma of the median nerve. Arch Iran Med 2010;13:45-7.  Back to cited text no. 13
    
14.
Sondergaard G, Mikkelsen S. Fibrolipomatous hamartoma of the median nerve. J Hand Surg Br 1987;12:224-5.  Back to cited text no. 14
    
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Senger JL, Classen D, Bruce G, Kanthan R. 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. Plast Surg (Oakv) 2014;22:201-6.  Back to cited text no. 15
    
16.
Hauck RM, Banducci DR. The natural history of a lipofibromatous hamartoma of the palm: A case report. J Hand Surg Am 1993;18:1029-31.  Back to cited text no. 16
    
17.
Bisceglia M, Vigilante E, Ben-Dor D. Neural lipofibromatous hamartoma: A report of two cases and review of the literature. Adv Anat Pathol 2007;14:46-52.  Back to cited text no. 17
    
18.
Tahiri Y, Xu L, Kanevsky J, Luc M. Lipofibromatous hamartoma of the median nerve: A comprehensive review and systematic approach to evaluation, diagnosis, and treatment. J Hand Surg Am 2013;38:2055-67.  Back to cited text no. 18
    
19.
Paletta FX, Senay LC Jr. Lipofibromatous hamartoma of median nerve and ulnar nerve: Surgical treatment. Plast Reconstr Surg 1981;68:915-21.  Back to cited text no. 19
    
20.
Houpt P, Storm van Leeuwen JB, van den Bergen HA. Intraneural lipofibroma of the median nerve. J Hand Surg Am 1989;14:706-9.  Back to cited text no. 20
    
21.
Bergman FO, Blom SE, Stenström SJ. Radical excision of a fibro-fatty proliferation of the median nerve, with no neurological loss symptoms. Plast Reconstr Surg 1970;46:375-80.  Back to cited text no. 21
    
22.
Friedlander HL, Rosenberg NJ, Graubard DJ. Intraneural lipoma of the median nerve. Report of two cases and review of the literature. J Bone Joint Surg Am 1969;51:352-62.  Back to cited text no. 22
    
23.
Rowland SA. Lipofibroma of the median nerve in the palm. J Bone Joint Surg Am 1967;49:1309-13.  Back to cited text no. 23
    
24.
Terzis JK, Daniel RK, Williams HB, Spencer PS. Benign fatty tumors of the peripheral nerves. Ann Plast Surg 1978;1:193-216.  Back to cited text no. 24
    
25.
Patil VS, Nagle S. Lipofibromatous hamartoma of the median nerve: A case report and review of the literature. Indian J Plast Surg 2009;42:122-5.  Back to cited text no. 25
[PUBMED]  [Full text]  
26.
Afshar A, Assadzadeh O, Mohammadi A. Ultrasonographic diagnosis of lipofibromatous hamartoma of the median nerve. Iran J Radiol 2015;12:e11270.  Back to cited text no. 26
    
27.
Mert M, Hacısalihoglu P. Lipofibromatous hamartoma of the plantar nerve an extremely rare localization. J Am Podiatr Med Assoc 2018;108:182-5.  Back to cited text no. 27
    
28.
Abdelshaheed ME. Extensive intraneural fascicular dissection of a lipofibromatous hamartoma of the ulnar digital nerve of the thumb. Eur J Plast Surg 2018;41:605-8.  Back to cited text no. 28
    
29.
Callison JR, Thoms OJ, White WL. Fibro-fatty proliferation of the median nerve. Plast Reconstr Surg 1968;42:403-13.  Back to cited text no. 29
    
30.
Jain TP, Srivastava DN, Mittal R, Gamanagatti S. Fibrolipomatous hamartoma of median nerve, Australas Radiol Journal 2007; Spec number: B98-B100.  Back to cited text no. 30
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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