ADULT-ONSET CYSTIC HYGROMA IN AXILLA: A RARE CASE REPORT FROM INDIA AND LITERATURE REVIEW

(case report)

Authors

DOI:

https://doi.org/10.11603/ijmmr.2413-6077.2021.1.12093

Keywords:

cystic hygroma, cystic lymphangioma, adult-onset, axilla

Abstract

Background. Cystic hygroma (CH), occurs in 1/6000 live births and in 90% of cases develops in age less than 2 years old. They are mainly located in cervicofacial region. Adult-onset CH is very rare.

Objective. The aim of this study is to review literature to discuss the clinical presentation, diagnosis, and treatment of CH in adults through a case report of unilocular CH in the axillary region in an adult male from India.

Methods. A first case report of unilocular CH in axillary region in an adult male from India is being investigated.

Results. Here we report a case of unilocular CH in the axillary region in a 49-year-old male with a 14x16x8 cm cystic swelling in left axilla with a history of aspiration failure. Contrast-enhanced MRI (CEMRI) showed well-defined thin walled, unilocular cystic lesion which appeared hyperintense on T2 & STIR and hypointense on T1W1 and showed thin peripheral rim of enhancement on post contrast images. The patient underwent surgical excision and the diagnosis of a pathological CH was established. His postoperative recovery was uneventful and had no evidence of recurrence.

Conclusion. Due to rarity of adult-onset unilocular CH in axilla, its evaluation for prompt diagnosis and definitive treatment to prevent recurrence and complications is urgent. Furthermore, this is the first reported case from India which has been successfully managed at a peripheral hospital in Northeast-India and our report of this case contributes to the evidences supporting the role of CH in a differential diagnosis for masses in the adult axilla, especially in acute phase with no predisposing factors.

Author Biographies

R. Pandey, BASE HOSPITAL, TEZPUR, ASSAM, INDIA

Assistant Professor Surgery, Department of Surgery, Base Hospital, Tezpur, Assam, India

Ranjan Kumar, BASE HOSPITAL, TEZPUR, ASSAM, INDIA

Assistant Professor Surgery, Department of Surgery, Base Hospital, Tezpur, Assam, India

S. Maheshwari, BASE HOSPITAL, TEZPUR, ASSAM, INDIA

Assistant Professor Radiology, Department of Radiology, Base Hospital, Tezpur, Assam, India

T. S. Singh, BASE HOSPITAL, TEZPUR, ASSAM, INDIA

Assistant Professor Pathology, Department of Radiology, Base Hospital, Tezpur, Assam, India

S. Bhalla, BASE HOSPITAL, TEZPUR, ASSAM, INDIA

Associate Professor Clinical Microbiology and Infectious Diseases, Command Hospital, Udhampur, India

I. D. Khan, COMMAND HOSPITAL, UDHAMPUR, INDIA

Associate Professor Clinical Microbiology and Infectious Diseases, Command Hospital, Udhampur, India

References

Akar E. Various anatomic localizations of cystic hygroma: a retrospective analysis of 16 cases. The Turkish Journal of Thoracic and Cardiovascular Surgery 2017;25:242–248.

DOI: 10.5606/tgkdc.dergisi.2017.13439

Lodhia J, Philemon R, Amsi P, et al. A Report of a Large Axillary Cystic Hygroma (a.k.a Lymphan­gioma) in a Newborn from a Tertiary Hospital in Northern Tanzania. Case Rep Surg 2020;2020: 5624019–5624019.

DOI: 10.1155/2020/5624019

Mirza B, Ijaz L, Saleem M, et al. Cystic hygroma: an overview. J Cutan Aesthet Surg 2010; 3: 139–144.

DOI: 10.4103/0974-2077.74488

McCaffrey F, Taddeo J. Surgical management of adult-onset cystic hygroma in the axilla. Int J Surg Case Rep 2014;7:29–31.

DOI: 10.1016/j.ijscr.2014.11.017

Güner A, Aydin A, Çeli̇k F. Cystic hygromas in adults: Reports of two cases. Bakırköy Tıp Dergisi 2006;2:101–103.

Smith RC, Sherk HH, Kollmer C, et al. Cystic lymphangioma in the adult: An unusual axillary mass. Magnetic Resonance Imaging 1989; 7: 561–563.

DOI: 10.1016/0730-725X(89)90411-6

Michail O, Michail P, Kyriaki D, et al. Rapid Development of an Axillary Mass in an Adult: A Case of Cystic Hygroma: Southern Medical Journal 2007; 100:845–849.

DOI: 10.1097/SMJ.0b013e3180f60e09

Mansingani S, Desai N, Pancholi A, et al. A case of axillary cystic hygroma. Indian Journal of Radiology and Imaging 2005;15:517.

Veeraraghavan G, Denny C, Lingappa A. Cystic hygroma in an adult; a case report. Libyan J Med 2009; 4:160–161.

DOI: 10.4176/090608

Bahl S, Shah V, Anchlia S, et al. Adult-onset cystic hygroma: A case report of rare entity. Indian J Dent 2016;7:51–54.

DOI: 10.4103/0975-962X.179374

Shukla S, Shivhare R, Lal V, et al. Cystic lym­phangioma of breast and axillary region in an adult: a rare presentation. International Surgery Journal 2020;8:391–394.

DOI: 10.18203/2349-2902.isj20205911

Manikoth P, Mangalore G, Megha V. Axillary Cystic Hygroma. Journal of Postgraduate Medicine 2004;50:215–216.

Karabulut D, Kula O, Ustabasioglu FE, et al. Imaging findings of axillary cystic lymphangioma in adult. Diagnostic and Interventional Imaging 2018;99: 111–113.

DOI: 10.1016/j.diii.2017.06.007

Colangeli W, Facchini V, Kapitonov A, et al. Cystic lymphangioma in adult: a case report and a review of the literature. Journal of Surgical Case Reports; 2020. Epub ahead of print 1 July 2020.

DOI: 10.1093/jscr/rjaa179

Loetscher KCQ, Jandali A-R, Garzoli E, et al. Axillary Cavernous Lymphangioma in Pregnancy and Puerperium. GOI 2005;60:108–111.

DOI: 10.1159/000085584

Lian HH, Sha PP. Late- Onset Cystic Hygromas Presenting in an Adult: A Case Report. 2016; 3.

Schefter RP, Olsen KD, Gaffey TA. Cervical Lymphangioma in the Adult. Otolaryngol Head Neck SuRG 1985;93:65–69.

DOI: 10.1177/019459988509300113

Chotai N, Fok E, Chan P, et al. Axillary lym­phangioma in an asymptomatic adult female. The Breast Journal 2018;24:415–416.

DOI: 10.1111/tbj.12915

Elshaar K, AbuAleid L. Adult-onset giant cervical cystic hygroma with pressure manifestations on aerodigestive tract, managed surgically: repor­ting of a rare case. Ann R Coll Surg Engl 2019;101: e84–e87.

DOI: 10.1308/rcsann.2018.0214

Nguyen K, Karsif K, Lee S, et al. Lymphangioma in an Elderly Patient: An Unusual Cause of Axillary Mass. The Breast Journal 2011;17:416–417.

DOI: 10.1111/j.1524-4741.2011.01103.x

Naidu SI, McCalla MR. Lymphatic malfor­ma­tions of the head and neck in adults: a case report and review of the literature. Ann Otol Rhinol Laryngol 2004;113:218–222.

DOI: 10.1177/000348940411300309

Suk S, Sheridan M, Saenger JS. Adult lym­phangioma: a case report. Ear Nose Throat J 1997;76: 881–883.

PMID: 9431778

Park T, Lee HS, Jung EJ, et al. Concomitant breast and axillary lymphangioma in an adult: A case report and a review of the literature. Medicine; 97, https://journals.lww.com/md-journal/Fulltext/ 2018/ 11090/Concomitant_breast_and_axillary_lymphan­gio­ma_in_an.9.aspx (2018).

Adams MT, Saltzman BS, Perkins JA. Treatment of Lymphatic Malformations: A Systematic Review. Otolaryngol Head Neck Surg 2011;145:P249–P249.

DOI: 10.1177/0194599811415823a374

Adams MT, Saltzman B, Perkins JA. Head and Neck Lymphatic Malformation Treatment: A Syste­matic Review. Otolaryngol Head Neck Surg 2012;147: 627–639.

DOI: 10.1177/0194599812453552

Figaro N, Rampersad R, Juman S. Doxycycline Sclerotherapy of a Cervical Cystic Hygroma: A Carib­bean Institution Experience. Case Reports in Otola­ryngology 2020;2020:9187205.

DOI: 10.1155/2020/9187205

Rahul SK, Kumar B, Bhatnagar A, et al. Unusual Site for Cystic Hygroma: A Single Centre Experience. J Cutan Aesthet Surg 2016;9:287–289.

DOI: 10.4103/0974-2077.197091

Gow L, Gulati R, Khan A, et al. Mihaimeed F. Adult onset cystic hygroma: a case report and review of management. Grand rounds.

Michail O, Michail P, Kyriaki D, et al. Rapid Development of an Axillary Mass in an Adult: A Case of Cystic Hygroma: Southern Medical Journal 2007; 100:845–849.

DOI: 10.1097/SMJ.0b013e3180f60e09

Copley PC, Ali L, Mirza S. Spontaneous lym­phocoele: an unusual cause of an axillary mass. Case Reports 2016;2016:bcr2015213088.

DOI: 10.1136/bcr-2015-213088

Manzini M, Schweiger C, Manica D, et al. Response to OK-432 sclerotherapy in the treatment of cervical lymphangioma with submucosal extension to the airway. Brazilian Journal of Otorhinolaryngology 2020;86:127–129.

DOI: 10.1016/j.bjorl.2016.04.016

Sherman BE, Kendall K. A unique case of the rapid onset of a large cystic hygroma in the adult. American Journal of Otolaryngology 2001;22:206–210.

DOI: 10.1053/ajot.2001.23430

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Published

2021-11-22

How to Cite

Pandey, R., Kumar, R., Maheshwari, S., Singh, T. S., Bhalla, S., & Khan, I. D. (2021). ADULT-ONSET CYSTIC HYGROMA IN AXILLA: A RARE CASE REPORT FROM INDIA AND LITERATURE REVIEW: (case report). International Journal of Medicine and Medical Research, 7(1), 74–80. https://doi.org/10.11603/ijmmr.2413-6077.2021.1.12093