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- W3047335079 abstract "Pisiform and hamate coalition, a rare congenital anomaly, is most often identified as an asymptomatic incidental finding on radiographs. Some patients have symptomatic coalition with ulnar-sided wrist pain or ulnar nerve neuropathy from ulnar nerve compression. Sports activities such as cycling and weight lifting can cause compression of the ulnar nerve in the hand. This report describes a case of a pisiform and hamate coalition in a 36-year-old man who reported decreased right-hand dexterity and right ulnar-sided wrist pain. The patient, an amateur weight lifter, developed right claw hand through repeated bench press training. Intraoperative findings revealed compression of the deep palmar branch of the ulnar nerve between the tendinous arch of the hypothenar muscles and pisiform and hamate coalition. Surgical resection of the tendinous arch and the enlarged hook of hamate relieved the claw hand deformity. Pisiform and hamate coalition, a rare congenital anomaly, is most often identified as an asymptomatic incidental finding on radiographs. Some patients have symptomatic coalition with ulnar-sided wrist pain or ulnar nerve neuropathy from ulnar nerve compression. Sports activities such as cycling and weight lifting can cause compression of the ulnar nerve in the hand. This report describes a case of a pisiform and hamate coalition in a 36-year-old man who reported decreased right-hand dexterity and right ulnar-sided wrist pain. The patient, an amateur weight lifter, developed right claw hand through repeated bench press training. Intraoperative findings revealed compression of the deep palmar branch of the ulnar nerve between the tendinous arch of the hypothenar muscles and pisiform and hamate coalition. Surgical resection of the tendinous arch and the enlarged hook of hamate relieved the claw hand deformity. Carpal coalition is a rare congenital anomaly that is mostly identified as an asymptomatic incidental finding on radiographs. Carpal coalition can be of 2 forms: an osseous coalition in which the carpals are united by an osseous block, or a nonosseous coalition by which the carpals are united by cartilage or fibrous tissue.1Burnett S.E. Hamate–pisiform coalition: morphology, clinical significance, and a simplified classification scheme for carpal coalition.Clin Anat. 2011; 24: 188-196Crossref PubMed Scopus (28) Google Scholar The most common coalition is the lunate–triquetrum coalition.2DeVilliers Minnaar A.B. Congenital fusion of the lunate and triquetral bones in the South African Bantu.J Bone Joint Surg Br. 1952; 34: 45-48Crossref PubMed Google Scholar,3Gottschalk M.B. Danilevich M. Gottschalk H.P. Carpal coalitions and metacarpal synostoses: a review.Hand (NY). 2016; 11: 271-277Crossref PubMed Scopus (10) Google Scholar Pisiform–hamate coalition is a rare coalition that was first reported by Cockshott.4Cockshott W.P. Pisiform hamate fusion.J Bone Joint Surg Am. 1969; 51: 778-780Crossref PubMed Scopus (30) Google Scholar Few reported cases have included symptomatic coalition with ulnar-sided wrist pain.5Inui A. Mifune Y. Nishimoto H. Niikura T. Kuroda R. A case of a painful coalition between pisiform and hamate.Case Reports Plast Surg Hand Surg. 2019; 6: 35-37Crossref PubMed Google Scholar,6Nolte I. Mauler F. Sánchez T. Bilateral ulnar-sided wrist pain due to pisiform–hamate coalition.Case Rep Orthop. 2019; 2019: 5891972PubMed Google Scholar Only one article in the literature described ulnar nerve neuropathy with ulnar nerve compression by a pisiform–hamate coalition.7Berkowitz A.R. Melone Jr., C.P. Belsky M.R. Pisiform–hamate coalition with ulnar neuropathy.J Hand Surg Am. 1992; 17: 657-662Abstract Full Text PDF PubMed Scopus (17) Google Scholar Sports activities such as cycling and weight lifting reportedly cause compression of the ulnar nerve in the hand.8Krivickas L.S. Wilbourn A.J. Sports and peripheral nerve injuries: report of 190 injuries evaluated in a single electromyography laboratory.Muscle Nerve. 1998; 21: 1092-1094Crossref PubMed Scopus (39) Google Scholar This report describes a case of pisiform–hamate coalition in a 36-year-old man with ulnar nerve neuropathy triggered by repetitive bench press training. To our knowledge, this report is the first to describe a pure motor deficit of ulnar nerve with pisiform–hamate coalition. A 36-year-old man was referred to our hospital because of decreased dexterity of the right hand and right ulnar-sided wrist pain. The patient had a history of hyperuricemia and hyperlipidemia, particularly fatty liver. He had no upper-extremity trauma or surgery. He had started a yearlong weight loss program of weight training with bench presses of 100 kg (220 lb). Physical examination revealed no cubitus varus or valgus deformity in the left elbow. Tinel test results were negative at the cubital tunnel and Guyon tunnel. He showed no disturbance of sensation on either the left forearm or left hand. Semmes–Weinstein monofilament test revealed no diminished sensation. The right hand showed mild clawing of the ring and little fingers, and atrophy of the dorsal and palmar interosseous muscles was found (Fig. 1). Grip and key pinch strength of the affected hand was 30 and 6.0 kg, respectively, whereas that of the unaffected hand was 40 and 9.0 kg, respectively. Froment sign was positive on the right hand. Anteroposterior radiographs of the right hand (Fig. 2) and computed tomography (CT) revealed an unusual hamate shape compared with the unaffected side on the sagittal reconstruction view: a large hook of hamate and pisiform (Fig. 3). We observed a type 1 coalition according to Minaar’s classification,2DeVilliers Minnaar A.B. Congenital fusion of the lunate and triquetral bones in the South African Bantu.J Bone Joint Surg Br. 1952; 34: 45-48Crossref PubMed Google Scholar which was characterized by an incomplete fusion resembling pseudarthrosis. Ultrasonographic examination (Fig. 4) indicated no space-occupying lesion. A nerve conduction examination performed at the initial visit indicated normal amplitude of the right first dorsal interosseous muscle motor nerve conduction velocity of the ulnar nerve and normal sensory nerve conduction velocity on the affected side. However, 4 months later, the amplitude of the right first dorsal interosseous muscle motor nerve conduction velocity of the ulnar nerve had declined. We speculated that repeated compression of the palm during weight training might have caused neuropathy of the deep palmar branch of the ulnar nerve. We advised the patient not to perform bench press exercises. We observed the motor deficit for 10 months with no improvement. For this reason, surgical exploration was performed. Under general anesthesia, Guyon canal was opened and the deep palmar branch of the ulnar nerve was released. The deep branch of the ulnar nerve was compressed between the tendinous arch of the hypothenar muscles from the volar side and the hypertrophied hook of hamate from the dorsal side (Fig. 5A, 5B). We resected the tendinous arch of the hypothenar muscles and the hypertrophied hook of hamate (Fig. 5A). The deep branch was released completely. One week after surgery, the mild claw hand had recovered (Fig. 6). At 10 months after the operation, grip strength and key pinch strength in the affected hand was 39 and 8.0 kg, respectively, compared with 41 and 10 kg, respectively, on the unaffected side. Electrophysiological examination revealed recovery of the amplitude of the motor nerve conduction velocity of the ulnar nerve of the right dorsal first interosseous muscles.Figure 2Anteroposterior plain radiograph of the right wrist showing unusual shape as a large hook of hamate and pisiform.View Large Image Figure ViewerDownload (PPT)Figure 3Sagittal reconstruction CT of the right wrist showing a type 1 pisiform–hamate coalition.View Large Image Figure ViewerDownload (PPT)Figure 4Axial view of ultrasonography showing no space-occupying lesions.View Large Image Figure ViewerDownload (PPT)Figure 5A Deep branch of the ulnar nerve compressed from the dorsal side by the tendinous arch of the hypothenar muscles (arrowhead). B Deep branch compressed from the dorsal side by the hypertrophied hook of hamate (arrowhead).View Large Image Figure ViewerDownload (PPT)Figure 6Right hand at 1 week after surgery showing recovery of the mild claw hand.View Large Image Figure ViewerDownload (PPT) The patient in this case report presented with ulnar-sided wrist pain and pure motor neuropathy attributable to a lesion of the deep branch of the ulnar nerve with mild clawing of the ring and little fingers. As far as we know, this description is the first in the literature for a pure motor deficit with pisiform–hamate coalition. Classification of the level of the lesion in ulnar neuropathies of the hand presented by Wu et al9Wu J.S. Morris J.D. Hogan G.R. Ulnar neuropathy at the wrist: case report and review of literature.Arch Phys Med Rehabil. 1985; 66: 785-788PubMed Google Scholar demonstrated that the patient in this report had a pure motor disturbance, type 3 ulnar nerve neuropathy, as indicated by a lesion of the deep branch of the ulnar nerve just distal to the superficial branch but proximal to the branch to the hypothenars. For this patient, we speculate that the ulnar-side wrist pain derived from neuropathic pain. We identified the motor nerve disturbance as probably resulting from entrapment of the deep branch of the ulnar nerve. Krivickas and Wilbourn8Krivickas L.S. Wilbourn A.J. Sports and peripheral nerve injuries: report of 190 injuries evaluated in a single electromyography laboratory.Muscle Nerve. 1998; 21: 1092-1094Crossref PubMed Scopus (39) Google Scholar reported that sports activities such as weight lifting, and specifically bench press lifting, cause entrapment neuropathy as carpal tunnel syndrome and ulnar nerve neuropathy. In this case, we suspected that repetitive compression to the hand involved in bench press lifting contributed to the development of compression of the deep palmar branch of the ulnar nerve. Preoperative CT provided information related to pisiform–hamate coalition that was not initially recognized by plain radiographs. In fact, pisiform–hamate coalition is a rare coalition. An incidence of 0.11% to 0.76%1Burnett S.E. Hamate–pisiform coalition: morphology, clinical significance, and a simplified classification scheme for carpal coalition.Clin Anat. 2011; 24: 188-196Crossref PubMed Scopus (28) Google Scholar is estimated for this rare coalition, which is inferred as resulting from metaplasia of the pisohamate ligament into bone.1Burnett S.E. Hamate–pisiform coalition: morphology, clinical significance, and a simplified classification scheme for carpal coalition.Clin Anat. 2011; 24: 188-196Crossref PubMed Scopus (28) Google Scholar The patient in this case report presented the coalition categorized as type 1 by Minaar’s classification: incomplete fusion resembling pseudoarthrosis. The classification was developed by Minaar to classify lunotriquetral fusions based on radiographs.2DeVilliers Minnaar A.B. Congenital fusion of the lunate and triquetral bones in the South African Bantu.J Bone Joint Surg Br. 1952; 34: 45-48Crossref PubMed Google Scholar Berkowitz et al7Berkowitz A.R. Melone Jr., C.P. Belsky M.R. Pisiform–hamate coalition with ulnar neuropathy.J Hand Surg Am. 1992; 17: 657-662Abstract Full Text PDF PubMed Scopus (17) Google Scholar demonstrated 2 cases of pisiform–hamate coalition with mixed motor and sensory neuropathy of ulnar nerve. Two cases were type 1 coalition, which involved the floor of Guyon canal and compression of the ulnar nerve. Total excision of the pisiform and partial resection of the articulating portion of the hook of hamate decompressed the ulnar nerve and relieved the symptoms. In the patient in the current case report, the deep branch of the ulnar nerve was compressed between the tendinous arch of the hypothenar muscles and the hypertrophied hook of hamate. We resected the tendinous arch of the hypothenar muscles and the hypertrophied hook of hamate, rather than the pisiform, and decompressed the deep branch of the ulnar nerve completely. We believe that surgical procedures used for this coalition with ulnar nerve neuropathy should be chosen depending on the characteristics of the patient’s pathophysiology. Awareness of this pathophysiology can raise confidence in the proper treatment and surgical management of this rare condition, whereas delayed diagnosis and treatment may lead to functional damage.10Witt J.C. Stevens J.C. Neurologic disorders masquerading as carpal tunnel syndrome: 12 cases of failed carpal tunnel release.Mayo Clin Proc. 2000; 75: 409-413Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar Indeed, surgical resection of the hook of hamate and the tendinous arch resolved the pure motor neuropathy of the deep branch of the ulnar nerve. Surgeons should be aware that repeated compression motion to the palm such as that inherent in excessive weight training can cause ulnar nerve neuropathy in patients with pisiform–hamate coalition. ErrataJournal of Hand Surgery Global OnlineVol. 3Issue 6PreviewIn the article by Demino et al in the September 2020 issue of Journal of Hand Surgery Global Online (“Diagnostic Value of Ultrasound in CTS in Diabetic Versus Nondiabetic Populations”, Vol. 2, No. 5, p 267-271), the written informed consent statement was not included. The statement now appears here as follows: Written informed consent was obtained from the patient for publication of this case report and accompanying images. Full-Text PDF Open Access" @default.
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- W3047335079 title "Pisiform–Hamate Coalition With Entrapment Neuropathy of the Deep Palmar Branch of the Ulnar Nerve" @default.
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