10.5826/dpc.1401a54
text/xml
2160-9381
3607
en
Vincenzo Piccolo
https://creativecommons.org/licenses/by-nc/4.0
Dermatology Practical & Conceptual
scar
Cutaneous Bronchogenic Cyst Presenting as a Keloid on the Back – A Case Report
Text.Serial.Journal
https://dpcj.org/index.php/dpc
Research Letter
2024-01-31
2023-06-14
2024-01-31
2024-02-21
e2024054
Dermatol Pract Concept
Vincenzo Piccolo
2024/01/31
10.5826/dpc.1401a54
e2024054
Dermatology Unit, University of Campania, Naples, Italy
2160-9381
scar
en
e2024054
Tresser NJ, Dahms B, Berner JJ. Cutaneous bronchogenic cyst of the back: a case report and review of the literature. Pediatr Pathol. 1994;14(2):207-212. DOI: 10.3109/15513819409024254. PMID: 8008684.
Cutaneous Bronchogenic Cyst Presenting as a Keloid on the Back – A Case Report
https://dpcj.org/index.php/dpc/article/download/3607/2532
              Cutaneous Bronchogenic Cyst Presenting
              as a Keloid on the Back – A Case Report
    Julia Nowowiejska1, Camilo Arias-Rodriguez2, Giuseppe Argenziano3, Andrea Ronchi4,
                                     Vincenzo Piccolo3


1 Department of Dermatology and Venereology, Medical University of Bialystok, Poland
2 Department of Dermatology, Universidad Pontificia Bolivariana, Medellín, Colombia
3 Dermatology Unit, University of Campania, Naples, Italy
4 Pathology Unit, University of Campania, Naples, Italy



Key words: bronchogenic cyst, keloid, scar, dermoscopy, dermatopahtology
Citation: Nowowiejska J, Arias-Rodriguez C, Argenziano G, Ronchi A, Piccolo V. Cutaneous Bronchogenic Cyst Presenting as a Keloid on
the Back – A Case Report. Dermatol Pract Concept. 2024;14(1):e2024054. DOI: https://doi.org/10.5826/dpc.1401a54
Accepted: September 17, 2023; Published: January 2024
Copyright: ©2024 Nowowiejska 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: Julia Nowowiejska, MD, PhD, Department of Dermatology and Venereology, Medical University of Bialystok,
Poland. Phone: +48857409572 E-mail: julia.nowowiejska@umb.edu.pl




Introduction                                                             Case Presentation
Bronchogenic cyst (BC) is a rare congenital condition that               Young girl, Fitzpatrick III, first presented to the dermatol-
occurs due to an impaired process of lung development. The               ogy out-patient department at the age of 9 years. She had
pathogenesis involves a set of cells that separate from the              already undergone previously two surgeries due to a small
rest, which differentiate by themselves and develop a small              tumefaction, draining seropurulent exudation, located in the
lung bud [1]. BC is usually diagnosed in the first years of              scapular region. The excised tissue was sent for histopatho-
life and manifests clinically as a tumorous mass or orifice              logical examination and revealed a sebaceous cyst. The par-
draining fluid [2], symptomatic in about half of cases [1].              ents decided to visit a doctor again because, despite those
The treatment of choice is surgical removal [2].                         two surgeries, they were concerned about the persistent and
                                                                         continuous drainage from a small orifice on the surface of
                                                                         a lesion that resembled a keloid scar, which was addition-
                                                                         ally constantly growing (Figure 1A). Upon dermoscopy, the




Research Letter | Dermatol Pract Concept. 2024;14(1):e2024054                                                                         1
lesion displayed linear vessels arranged peripherally over a           development from multipotent stem cells that would un-
pink to white structureless background, resembling a ke-               dergo cystic degeneration [4].
loid (Figure 1B). On the latest evaluation, dermoscopy of                  The usual extrapulmonary localization of bronchogenic
the grown lesion additionally showed pigmented tan lines               cysts is mediastinum [1,2,4] however, in our case, it was
and circles arranged linearly, located also peripherally, which        the skin of the scapular region. In the available literature,
could correspond to pigmentation due to melanocyte acti-               there are only 15 cases of scapular BC, of which most were
vation due to scar formation. The patient’s parents refused            children with male predominance [2,3,5,6], unlike our case,
another excision or biopsy of the lesion at that time. Later,          however with no dermoscopy of this disorder. We studied all
the patient presented for the second time one year later due           available cases of BCs and the whole list is presented in the
to further enlargement of the lesion, but this time a deep             supplementary Table 1.
shaving biopsy was taken to exclude dermatofibrosarcoma                    In the described patient, the clinical picture involved a
protuberans. The result of the histopathological examination           slowly growing tumor with pus leakage, which is the usual
(Figure 1, C and D) unexpectedly revealed cutaneous bron-              presentation of BC [2]. However, later it resembled a keloid,
chogenic cyst.                                                         which was symptomatic and grew over time, hence the differ-
                                                                       ential diagnosis was dermatofibrosarcoma protuberans. We

Conclusions                                                            did not find any other BC description clinically resembling a
                                                                       keloid. We also need to take into account that it might have
Presumably, extrapulmonary BC located on the periscapu-                been actually a regular keloid scar after previous excisions
lar area develops due to an abnormal budding of the tra-               coexisting with a BC but hence, one must be careful in order
cheobronchial primitive foregut during early embryogenesis,            not to miss another pathology hidden underneath.
with migration to the thorax. Other proposed theories in-                  Possible complications include cyst rupturing or its
clude: metaplasia of pre-existing cutaneous tissue, primary            compression on other structures. In our case, none of
bizarre differentiation of skin cells in formation [3], and            these happened because it was located externally. Another




Figure 1. (A) Clinical picture of the bronchogenic cyst resembling a keloid scar. (B) Dermoscopic picture of the bronchogenic cyst; linear
vessels arranged peripherally over a pink to white structureless background, resembling a keloid. (C,D) Microscopic pictures of the sample
taken from the bronchogenic cyst, H&E, 10x and 40x, respectively.



2                                                                          Research Letter | Dermatol Pract Concept. 2024;14(1):e2024054
potential complication is infection. Noteworthy, it has been              ­literature. Pediatr Surg Int. 2005;21(10):843-845. DOI: 10.1007
documented that BC may undergo malignant transforma-                       /s00383-005-1531-5. PMID: 16180006.
                                                                     3.    Al-Balushi Z, Ehsan MT, Al Sajee D, Al Riyami M. Scapular
tion [1].
                                                                           Bronchogenic Cyst: A Case Report and Literature Review. Oman
   Hereby we describe a very rare case of cutaneous bron-
                                                                           Med J. 2012;27(2):161-163. DOI: 10.5001/omj.2012.33. PMID:
chogenic cyst. Considering the possibility of malignant trans-             22496944. PMCID: PMC3321343.
formation, dermatologists should be aware of such entities           4.    Aldawood Z, Moyer DJ, Woo SB. A Bronchogenic Cyst Masquer-
to early diagnose and remove the cyst.                                     ading as a Tongue Mass. Head Neck Pathol. 2021;15(4):1404-
                                                                           1408. DOI: 10.1007/s12105-021-01303-x. PMID: 33638760.
                                                                           PMCID: PMC8633169.
References                                                           5.    van der Putte SC, Toonstra J. Cutaneous 'bronchogenic' cyst.
                                                                           J Cutan Pathol. 1985;12(5):404-409. DOI: 10.1111/j.1600
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                                                                           -0560.1985.tb00437.x. PMID: 4056165.
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                                                                     6.    Tresser NJ, Dahms B, Berner JJ. Cutaneous bronchogenic cyst of
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                                                                           1994;14(2):207-212. DOI: 10.3109/15513819409024254.
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Research Letter | Dermatol Pract Concept. 2024;14(1):e2024054                                                                                3
