Fibrous Hamartoma of Infancy: A Firm Plaque Presenting with Hypertrichosis and Hyperhidrosis

Melnick, Laura; Berger, Emily; Elenitsas, Rosalie; Chachkin, Samuel; Treat, James R.

doi:10.1111/pde.12459

Cited by 8 publications

(4 citation statements)

References 14 publications

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“…First, Weinberger et al 7 reported a case of FHI presenting as a perspiring neck mass in 1993. Next, in 2015, Melnick et al 6 reported a case of FHI presenting with hypertrichosis and hyperhidrosis, which is quite similar to our case. Both cases were reported in the US, and no case of FHI presenting with hyperhidrosis has been reported in Korea, until now.…”

Section: Discussionsupporting

confidence: 88%

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The First Reported Case of Fibrous Hamartoma of Infancy with Hyperhidrosis and Hypertrichosis in Korea

et al. 2018

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Fibrous hamartoma of infancy (FHI) is a rare entity with a benign nature. The typical clinical features are a single, slowly growing, painless mass on the trunk that appears within the first 2 years of life. We report a 13-month-old boy who presented with a plaque on the lower back since 4 months of age. The plaque had gradually become larger and firm, and hyperhidrosis and hypertrichosis were noticed. No visible connection between the spinal cord and the lesion was found in radiologic studies, indicating a disease other than spinal dysraphism. Histopathological findings showed well-defined fibrous trabeculae, mature adipose tissue, and primitive mesenchymal cells, all consistent with FHI. This is the first case of FHI presenting with hyperhidrosis and hypertrichosis reported in Korea.

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Section: Discussionsupporting

confidence: 88%

“…However, a review of the literature reveals only 2 cases of FHI presenting with hyperhidrosis in the world, and no case in Korea. 6 7 In this study, we report the first Korean case of FHI with hyperhidrosis and hypertrichosis.…”

Section: Introductionmentioning

confidence: 93%

The First Reported Case of Fibrous Hamartoma of Infancy with Hyperhidrosis and Hypertrichosis in Korea

et al. 2018

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“…Its pathogenesis and biological characteristics are still unclear. The disease usually occurs in children less than two years of age, of which approximately 23% are born with it, and male to female incidence ratio is approximately 2.4[ 5 ]. FHI mostly occurs in the axilla, followed by the upper arm, thigh, back, groin, buttocks, and external genitalia[ 4 - 6 ].…”

Section: Discussionmentioning

confidence: 99%

Fibrous hamartoma of infancy with bone destruction of the tibia: A case report

Qiao¹,

Yang²,

Chang³

et al. 2022

World J Clin Cases

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BACKGROUND Fibrous hamartoma of infancy (FHI) is a rare disease of infancy with unknown etiology. The disease mainly involves soft tissue, has no specific clinical manifestations, and is difficult to diagnose. At present, the diagnosis is mainly confirmed by histopathological examination, and the main treatment is surgical resection of the pathological tissue, which is prone to recurrence. CASE SUMMARY A five-month-old female patient was admitted to our hospital with swelling in the right calf. Two biopsies were performed in our hospital and another hospital, respectively, confirming the diagnosis as fibrous hamartoma. After exclusion of surgical contraindications, resection was performed with clear margins of 1 cm. Radiographic examination showed tumor recurrence more than four months after the operation, and surgery was performed again to extend the resection margins to 1.5 cm. The patient is recovering well, and after a follow-up of 36 mo, shows no signs of recurrence. CONCLUSION Our case report demonstrates that FHI should be considered in the differential diagnosis for a lower extremity mass with bone destruction. For FHI with bone destruction and unclear boundaries, excision margins of 1.5 cm could be superior to margins of 1 cm.

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“…244,245 The overlying skin is usually unremarkable with a few reported exceptions. 246,247 In a minority of cases, the tumor may be noted at birth. Most tumors measure less than 5 cm with the exceptional case greater than 10 cm.…”

Section: Fibrous and Myofibroblastic Tumorsmentioning

confidence: 99%

Nonepithelial Tumors and Tumor-like Lesions of the Skin and Subcutis in Children

Dehner

Gru

2018

Pediatr Dev Pathol

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This overview of mesenchymal tumors presenting in the skin and/or subcutis in children brings together the range of neoplasms and hamartomas which are seen in this age-group. It is not surprising from the perspective of the pediatric or general surgical pathologist that vascular anomalies, including true neoplasms and vascular malformations, are the common phenotypic category. Since there is considerable morphologic overlap among these lesions, clinicopathologic correlation may be more important than for many of the other mesenchymal tumors. The skin and subcutis are the most common sites of clinical presentation for the infantile myofibroma which is the most common of fibrous mesenchymal tumors in children. Several of the other mesenchymal tumors are more common adults-like dermatofibrosarcoma protuberans, but nonetheless have an important presence in children, even as a congenital neoplasm. A lipomatous tumor in a young child should be considered as a possible manifestation of an overgrowth syndrome.

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Fibrous Hamartoma of Infancy: A Firm Plaque Presenting with Hypertrichosis and Hyperhidrosis

Cited by 8 publications

References 14 publications

The First Reported Case of Fibrous Hamartoma of Infancy with Hyperhidrosis and Hypertrichosis in Korea

The First Reported Case of Fibrous Hamartoma of Infancy with Hyperhidrosis and Hypertrichosis in Korea

Fibrous hamartoma of infancy with bone destruction of the tibia: A case report

Nonepithelial Tumors and Tumor-like Lesions of the Skin and Subcutis in Children

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