10.5826/dpc.1304a206
text/xml
2160-9381
3423
en
Niti  Khunger
https://creativecommons.org/licenses/by-nc/4.0
Dermatology Practical & Conceptual
Sudden-Onset Pain in Fibrous Hamartoma of Infancy in a Child: An Atypical Presentation
Text.Serial.Journal
https://dpcj.org/index.php/dpc
Image Letter
2023-10-31
2023-04-11
2023-10-31
2024-01-15
e2023206
Dermatol Pract Concept
Niti  Khunger
2023/10/31
10.5826/dpc.1304a206
e2023206
Department of Dermatology & STD, Vardhman Mahavir Medical College & Safdarjung Hospital, New Delhi, India
2160-9381
en
e2023206
with sarcomatous features. Mod Pathol. 2017;30(4):474-485. DOI: 10.1038/modpathol.2016.215. PMID: 28059097.
Sudden-Onset Pain in Fibrous Hamartoma of Infancy in a Child: An Atypical Presentation
https://dpcj.org/index.php/dpc/article/download/3423/2367
          Sudden-Onset Pain in Fibrous Hamartoma
        of Infancy in a Child: An Atypical Presentation
                     Prabin P. Dev 1, Prashant Verma1, Shruti Sharma2, Niti Khunger1


1 Department of Dermatology & STD, Vardhman Mahavir Medical College & Safdarjung Hospital, New Delhi, India
2 National Institute of Pathology, Indian Council of Medical Research, Department of Health Research, Ministry of Health and Family
  Welfare, Govt. of India, New Delhi, India



Citation: Dev PP, Verma P, Sharma S, Khunger N. Sudden-Onset Pain In Fibrous Hamartoma Of Infancy In A Child: An Atypical
Presentation. Dermatol Pract Concept. 2023;13(4):e2023206. DOI: https://doi.org/10.5826/dpc.1304a206
Accepted: May 5, 2023; Published: October 2023
Copyright: ©2023 Dev et al. This is an open-access article distributed under the terms of the Creative Commons Attribution-
NonCommercial License (BY-NC-4.0), https://creativecommons.org/licenses/by-nc/4.0/, which permits unrestricted noncommercial use,
distribution, and reproduction in any medium, provided the original authors and source are credited.
Funding: None.
Competing Interests: None.
Authorship: All authors have contributed significantly to this publication.
Corresponding Author: Prabin P. Dev; Department of Dermatology & STD, Vardhman Mahavir Medical College & Safdarjung Hospital,
K-16, Green Park extension, South Delhi, India; e-mail- drprabindev@gmail.com; Telephone: 9496669799




Case Presentation                                                        the mass followed by split skin graft closure and is now under
                                                                         regular follow-up with no recurrence since last 6 months.
A 10-year-old male child presented with a slowly progressive
plaque on his right posterior thigh that had been increasing
                                                                         Teaching Point
in size over the last 9 years, and which turned painful in the
last 2 weeks. On examination, an ill-defined plaque mea-                 FHI is a rare fibroproliferative lesion usually occurring within
suring 4 cm x3 cm with a hyper-pigmented, nodular surface                first two years of life with higher predilection in males [1].
was observed (Figure 1A). The lesion was firm, adherent to               FHI presents as a rapidly growing, firm mass of the subcutis
underlying structures, associated with tenderness and a local            in the axilla, shoulder, upper arm, inguinal region, or chest
rise in temperature. Biopsy revealed follicular plugging, low            wall [1]. Our patient had the lesion on the posterior thigh.
papillomatosis, mild perivascular chronic infiltrate, and thick          There are only three reported cases of FHI in the thigh [1]. The
benign interlacing fibrous bands with muscle bundles extend-             presentation of our patient was unique, as sudden onset of
ing into the adipose tissue in the lower dermis and subcutis             excruciating pain had not been previously reported. Although
(Figure 1, B and C). MRI revealed diffuse soft tissue swelling           histopathology did not reveal the underlying cause of the pain,
in the thigh. A diagnosis of fibrous hamartoma of infancy                we hypothesize that nerve entrapment or unnoticed trauma
(FHI) was made. The child underwent complete excision of                 leading to infection could be possible causes. Histopathology




Image Letter | Dermatol Pract Concept. 2023;13(4):e2023206                                                                             1
       Figure 1. (A) Ill-defined plaque measuring 4x3 cm with a hyper-pigmented, nodular surface on right posterior thigh. (B)
       ­Photomicrograph shows epidermis with low papillomatosis. Dermis appears sclerosed with mild chronic inflammation
        (H&E; 40X). (C) Photomicrograph shows deeper part of dermis and subcutis. Thick benign interlacing fibrous bands are
        seen extending into the subcutis (H&E; 40X).




shows characteristic triphasic pattern of fibro-collagenous tis-      References
sue, primitive mesenchymal cells, and mature fat [2]. Also, the
surface changes in the plaque seen in our patient correlated          1. Vinayak RS, Kumar S, Chandana S, Trivedi P. Fibrous hamar-
                                                                         toma of infancy. Indian Dermatol Online J. 2011;2(1):25-27.
with the papillomatosis on histopathology. Although FHI can
                                                                         DOI: 10.4103/2229-5178.79867. PMID: 23130211. PMCID:
be affixed to underlying tissue, it is usually benign. Very rarely,
                                                                         PMC3481792.
FHI can present with sarcomatous morphology [2]. Untreated            2. Al-Ibraheemi A, Martinez A, Weiss SW, et al. Fibrous hamartoma
cases, like in our patient, have the potential to continue grow-         of infancy: a clinicopathologic study of 145 cases, including 2
ing without regression. Local excision is the successful treat-          with sarcomatous features. Mod Pathol. 2017;30(4):474-485.
ment for FHI, with a low incidence of local recurrence.                  DOI: 10.1038/modpathol.2016.215. PMID: 28059097.




2                                                                           Image Letter | Dermatol Pract Concept. 2023;13(4):e2023206
