Milind S. Tullu Additional Professor, Department of Pediatrics, Seth G.S. Medical College & K.E.M. Hospital, Mumbai 400012, Maharashtra, India
Sidaraddi Sanju Senior Resident, Department of Pediatrics, Seth G.S. Medical College & K.E.M. Hospital, Mumbai 400012, Maharashtra, India
Rucha Tonge Junior Resident-II, Department of Pediatrics, Seth G.S. Medical College & K.E.M. Hospital, Mumbai 400012, Maharashtra, India
Mukesh Agrawal Ex-Professor & Head, Department of Pediatrics, Seth G.S. Medical College & K.E.M. Hospital, Mumbai 400012, Maharashtra, India
Address for correspondence: Milind S. Tullu, Additional Professor, Department of Pediatrics, Seth G.S. Medical College & K.E.M. Hospital, Mumbai 400012, Maharashtra, India E-mail: milindtullu@yahoo.com
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Sidaraddi Sanju, Milind S. Tullu, Rucha Tonge, et al./Duplication Cysts and Autoimmune Hemolytic Anemia: An Association
or a Coincidence?/Indian J Trauma Emerg Pediatr.2023;15(2):47–52.
Timeline
Received : April 03, 2023
Accepted : April 27, 2023
Published : June 30, 2023
Abstract
Duplication cysts are rare congenital anomalies of the gastrointestinal tract and the occurrence of multiple such cysts is an extremely rare occurrence. Autoimmune hemolytic anemia is an extremely unusual manifestation of these duplication cysts. Herein, we report a case with two duplication cysts (one cyst in the thorax; another in the abdomen)
complicated with autoimmune hemolytic anemia. This patient was a five year-old-male child who presented with congestive cardiac failure due to severe anemia (which, on evaluation, was diagnosed as autoimmune hemolytic anemia). Imaging studies (chest radiograph and computed tomography) revealed duplication cysts in the chest and abdomen. Anemia flare-up was noted in the patient despite two months of prednisolone therapy (2 mg/kg/day) and two high dose intravenous methylprednisolone pulse therapy courses (30 mg/kg/day for 5 days each). The steroid refractory autoimmune hemolytic anemia responded to rituximab (375 mg/m2 body surface area/week for 4 weeks). Four months after tapering off the steroid therapy, the patient did not experience flare-up of the anemia. To the best of our knowledge, this is the youngest and first pediatric age group patient to be reported, with a possible association between gastrointestinal duplication cysts with autoimmune hemolytic anemia.
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Data Sharing Statement
There are no additional data available. All raw data and code are available upon request.
Funding
This research received no funding.
Author Contributions
All authors contributed significantly to the work and approve its publication.
Ethics Declaration
This article does not involve any human or animal subjects, and therefore does not require ethics approval.
Acknowledgements
We would like to express our gratitude to the patients, their families, and all those who have contributed to this study.
Conflicts of Interest
No conflicts of interest in this work.
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Cite this article
Sidaraddi Sanju, Milind S. Tullu, Rucha Tonge, et al./Duplication Cysts and Autoimmune Hemolytic Anemia: An Association
or a Coincidence?/Indian J Trauma Emerg Pediatr.2023;15(2):47–52.
This license enables reusers to distribute, remix, adapt, and build upon the material in any medium or format for noncommercial purposes only, and only so long as attribution is given to the creator.
This license enables reusers to distribute, remix, adapt, and build upon the material in any medium or format for noncommercial purposes only, and only so long as attribution is given to the creator.
Description: Investigations in the patient on admission to our institution. (*Indicates abnormal values)
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Description: Frontal radiograph of chest showing left-sided rounded well-circumscribed radio-opaque mass without any internal calcifications or air fluid levels.
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Description: Computed tomography of the chest showing a well defined homogenously hypodense cystic lesion in the left paravertebral region
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Description: Computed tomography of the abdomen showing a cystic lesion anterior to the left kidney.
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Description: Graph showing the trend of haemoglobin (Hb), thin red arrows directed upwards indicate blood transfusions given, thick green arrows directed downwards indicate the intravenous methylprednisolone pulse doses administered, and the red stars indicate the intravenous rituximab therapy.