Melorheostosis: A case report

Alpoim, Bruno; Rodrigues, Maria Elisa; Félix, António; Marques, Pedro; Sá, Pedro; Silva, Luís Fernando Nunes Pires

doi:10.1016/j.rboe.2012.07.007

Cited by 4 publications

(5 citation statements)

References 4 publications

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“…Melorheostosis is a rare connective tissue disease characterized by sclerosing bone dysplasia. The incident is approximately 0.9 cases per million inhabitants [1] [6]. With about 400 cases of melorheostosis being reported in the literature up to date [7], the aetiology of the disease itself remains unknown.…”

Section: Discussionmentioning

confidence: 99%

Polyostotic Melorheostosis: Approach to Clinical Evaluation and Management

Teoh¹,

Teoh²,

Chieng³

et al. 2019

OJO

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Melorheostosis is a rare form of sclerosing bone dysplasia that involves mainly the long bone of the limbs. However, the involvement of more than one long bone, either involving ipsilaterally or contralaterally, is extremely rare. The aetiology still remains unknown despite approximately 400 cases have been reported in the literature up to date. Diagnosis is mainly by conventional radiographic imaging such as plain X-ray, in which the typical melting wax dripping down a candle appearance is seen. The main treatment options are conservative management involving mainly analgesias, physiotherapy, braces, and possible nerve block and symphathectomies if indicated. The aim of this case report is to discuss the approach to a case of melorheostosis involving two long bones of the ipsilateral lower limb (polyostotic) that presented to us in our centre. Appropriate investigations and treatment modalities must be tailored according to the patient's complaints in order to achieve satisfactory treatment outcomes. With adequate analgesia complimenting with appropriate physiotherapy and rehabilitation, our patient's symptoms improved dramatically.

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Section: Discussionmentioning

confidence: 99%

Polyostotic Melorheostosis: Approach to Clinical Evaluation and Management

Teoh¹,

Teoh²,

Chieng³

et al. 2019

OJO

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“…Until 2012 the etiology was not known, but the X-ray finding was given and well known. Also, the treatment of that patient was symptomatic without any surgical interventions [13].…”

Section: Discussionmentioning

confidence: 93%

A Rare Case of Melorheostosis in the Hand of a Saudi Woman

Abed

Mashrah

Almahdaly

et al. 2020

Cureus

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Melorheostosis is a very rare bone dysplasia, especially in the hand. Most cases were diagnosed incidentally, with the lower limbs being the most affected. This is the first Saudi woman with hand melorheostosis. A 33-year-old Saudi female had mild to moderate right-hand pain that started six years ago. Hand examination showed a full range of motion and full hand grip, and there was no tenderness upon palpation. Plain X-ray, unenhanced CT scan, and MRI of the hand showed an appearance resembling dripping candle wax as melorheostosis. The bone scan showed a nonvascular and nonacute lesion. An unenhanced CT scan demonstrated cortical and endosteal hyperostosis involving the proximal, middle, and distal third and fourth phalanges. Multisequential MRI of the hand demonstrated cortical hyperostosis involving the ulnar and radial aspect of the right fourth proximal, middle, and distal phalanges. Features in the X-ray, CT scan, bone scan, and MRI confirmed a diagnosis of melorheostosis with associated flexor tenosynovitis.

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“…In our case, the blood results were within normal limit. Histopathologically, findings are nonspecific and often show a mixture of mature and immature bone in a dense formation with increased trabecular bone [3]. Treatment is conservative for pain alleviation and physiotherapy although in severe cases, surgical intervention may be required, including tendon release, osteotomies, and even amputation.…”

Section: Discussionmentioning

confidence: 99%

“…The estimated incidence of melorheostosis is 1 in 1,000,000. Melorheostosis affects men and women in equal proportions with occurrence reported in child and adult populations [3].…”

Section: Introductionmentioning

confidence: 97%

Atypical presentation of melorheostosis with soft tissues involvement: a case report

Chia

Haron

Malek

2021

Egypt J Radiol Nucl Med

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Background Melorheostosis is a skeletal disorder giving rise to a dripping wax appearance. The exact cause is still unclear, and the diagnosis is always challenging due to its wide differential diagnoses. Soft tissue involvement of melorheostosis has been reported in previous literatures but it is a rare phenomenon. Case presentation A 10-year-old child with melorheostosis presented with hematuria. Ultrasonography (USG) identified a cystic lesion in the right hemipelvis adjacent to the urinary bladder which was initially regarded as an ovarian cyst. Computed tomography (CT) confirmed the cystic lesion as a fusiform right internal iliac artery aneurysm, as well as multiple right retroperitoneal and right lower limb capillary hemangiomas with uterine involvement. Hence, the final diagnosis was atypical melorheostosis with vascular malformations. The patient was managed conservatively with Sirolimus therapy for the vascular anomalies. Conclusion Albeit the rare involvement of soft tissues, careful search for vascular malformation is recommended in melorheostosis.

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Melorheostosis: A case report

Cited by 4 publications

References 4 publications

Polyostotic Melorheostosis: Approach to Clinical Evaluation and Management

Polyostotic Melorheostosis: Approach to Clinical Evaluation and Management

A Rare Case of Melorheostosis in the Hand of a Saudi Woman

Atypical presentation of melorheostosis with soft tissues involvement: a case report

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