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

You, Moon Hyung; Shin, Dong Hoon; Choi, Jong Soo; Kim, Byeong Su; Kim, Yeonwoong; Kim, JoonGoon

doi:10.3346/jkms.2018.33.e66

Cited by 9 publications

(7 citation statements)

References 13 publications

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“…In some other cases reported in the literature, however, the lesions could also be showed as predominant intensity of fibrous tissue traversed by disperse strands of fatty component or vice versa [8–10, 16–18]. The interspersed strands usually existed in a parallel fashion, or even in a whirling appearance as in our cases [10, 12–15, 18, 24]. For some cases, the adipose or the fibrous tissue might aggregate at the local area in the lesion [19–22].…”

Section: Discussionsupporting

confidence: 60%

“…According to the proportion of the different intensities similar to that of fatty tissue or muscle on CT or MR imaging, the authors classified the lesions as the balanced type or non-balanced type. In the balanced type, the proportion of the two components was basically equal [12–15, 19–22]. In the non-balanced type, however, it was not equal at all, with one of them being predominant and the other one subordinate [5, 8–10, 16–18, 23, 26].…”

Section: Discussionmentioning

confidence: 99%

“…Stensby et al (2014) [14] 9/M1.5ShoulderSubcutaneous layerPigmentation4.5MRBalancedIll-definedVascularity(MR)Moderately enhanced(MR)4. You et al(2018) [15] 13/M9Lumbar areaSubcutaneous layerHypertrichosis Hyperhidrosis4US MRBalancedIll-defined5. Guo et al(2011) [19] 120/M120Parapharyngeal spaceSnoring5CTBalancedWell-definedCalcification(CT)6.…”

Section: Discussionmentioning

confidence: 99%

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Fibrous hamartoma of infancy: radiologic features and literature review

Yang

Zhang

et al. 2019

BMC Musculoskelet Disord

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Background Fibrous hamartoma of infancy(FHI) is a rare benign lesion most frequently occurring within the first year of life. So far, just over 200 cases have been reported in the English literature, in which the radiologic findings of FHI have not been fully described. Herein, 2 adult cases of FHI receiving treatment in our hospital and the published cases searched on PubMed are reviewed, with the emphasis on the discussion of the spectrum of MR findings and their histologic correlation. Case presentation We present two adult cases who aged 47 years and 19 years with slow growing masses beginning from their childhood in the posterior craniocervical area. On CT and MR imaging, the tumours showed as the superficially located lesions with ill-defined margins that involved the subcutaneous layer and its underlying muscles. The size of the lesions were 21.3 × 16.7 × 16 cm in case 1 and 20.2 × 19.3 × 13.6 cm in case 2. The tumours demonstrated heterogeneous intensities/signals with the adipose tissue presenting as the disperse strands or small focus of fatty intensity/signal. Parallel or whirling appearance, and dilated vessels were delineated in the cases. Contrast enhancement was administered in case 1 and marked enhancement was found. Conclusions The usually observed manifestation of FHI on CT and/or MR imaging is the strands of adipose/fibrous intensities traversing the lesions, with the characteristic parallel or whirling appearance in some cases. The tumours with ill-defined margins have the tendency to involve the underlying muscles. Some fibroblastic and adipocytic tumours should be ruled out in differential diagnosis.

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Section: Discussionsupporting

confidence: 60%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Fibrous hamartoma of infancy: radiologic features and literature review

Yang

Zhang

et al. 2019

BMC Musculoskelet Disord

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“…Overlying hyperpigmentation, hyperhidrosis, and/or hypertrichosis have also been reported, but these signs are not presented in our case. [ 12 ] Most of the lesions are small and solitary, the size ranging from 0.5 to 9.0 cm. [ 3 – 12 ] The largest tumor diameter reported in the literature is 20 cm in a 7-year-old Nigerian girl.…”

Section: Discussionmentioning

confidence: 99%

“…[ 12 ] Most of the lesions are small and solitary, the size ranging from 0.5 to 9.0 cm. [ 3 – 12 ] The largest tumor diameter reported in the literature is 20 cm in a 7-year-old Nigerian girl. However, her diagnosis of FHI was questioned.…”

Section: Discussionmentioning

confidence: 99%

Giant fibrous hamartoma of infancy

Sun

Hui

et al. 2020

Medicine

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Rationale: Fibrous hamartoma of infancy (FHI) is a rare benign soft-tissue tumor in children with a triphasic organoid histologic appearance. Patient concerns: We here report a case with the largest FHI published so far. A 13-month-old boy with a rapidly growing tumor 45 cm in length and 69.3 cm in circumference of the left lower extremity was identified immediately at birth. Diagnoses: The diagnosis was uncertain before the operation, although biopsy was conducted. The postoperation histological examination showed arranged spindle cells, adipose tissue, and nests of immature small cells embedded in a myxoid background, which is the characteristic triphasic histology of FHI. Interventions: Under general anesthesia, hip and lower extremity amputation was performed. Outcomes: The patient was followed-up for 6 years and no signs of recurrence were found, suggesting a favorable prognosis, although a part of the residual tumor was present after the surgery. Lessons: FHI exhibits pathological and clinical characteristics. However, differential diagnosis of FHI from other soft-tissue tumors before operation remains a challenge. Thus, sometimes, aggressive therapy for the treatment of FHI might be a good choice to get a satisfactory.

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