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International Journal of Case Reports and Images (IJCRI)International Journal of Case Reports and Images (IJCRI) is an international, peer reviewed, monthly, open access, online journal, publishing high-quality, articles in all areas of basic medical sciences and clinical specialties.

Aim of IJCRI is to encourage the publication of new information by providing a platform for reporting of unique, unusual and rare cases which enhance understanding of disease process, its diagnosis, management and clinico-pathologic correlations.

IJCRI publishes Review Articles, Case Series, Case Reports, Case in Images, Clinical Images and Letters to Editor.

Website: www.ijcasereportsandimages.com

Reversible stomatocytosis

Akanksha Agrawal, Deepanshu Jain, Mitchell Goldstein

ABSTRACT

Abstract is not required for Clinical Images

(This page in not part of the published article.)

International Journal of Case Reports and Images, Vol. 8 No. 6 June 2017. ISSN – [0976-3198]

Int J Case Rep Images 2017;8(6):417–419. www.ijcasereportsandimages.com

Agrawal et al. 417

CASE REPORT OPEN ACCESS

Reversible stomatocytosis

Akanksha Agrawal, Deepanshu Jain, Mitchell Goldstein

CASE REPORT

Stomatocytosis is a rare morphological condition of the red blood cells in which the normal central zone of pallor is replaced by a mouth-like or slit-like pattern. Stomatocytosis can be either hereditary or acquired in the setting of acute alcohol consumption, chronic liver disease [1]. It may be asymptomatic or lead to hemolysis presenting as anemia. We present a case of a young lady with chronic alcoholism who presented with symptomatic anemia. Alcoholism is known to cause hypophosphatemia [2], which can cause stomatocytosis. Repletion of phosphorus and abstinence of alcohol improved the patient’s anemia, improving the stomatocytosis on the peripheral smear. The use of an easily available, rapid and cheap test like peripheral smear in guiding the therapy in such a patient reflects the importance of peripheral smear and its cost-effectiveness.

A 38-year-old African-American female with history of chronic pancreatitis, cholelithiasis, diabetes mellitus and alcohol abuse presented with severe anemia and light-headedness. She was consuming about 80 grams of alcohol on a daily basis. On admission, she had hemoglobin of 5.6 g/dL, mean corpuscular volume (MCV) 90 fL (normal), iron level 92 mg/dL (normal) and ferritin 1188 ng/mL. Her vitamin B12 and folate levels were normal and reticulocyte count corrected for anemia was 1.2%. Her phosphorus was 1.0 mg/dL (critically low). Abdominal ultrasound was consistent with hepatic

Akanksha Agrawal1, Deepanshu Jain1, Mitchell Goldstein2

Affiliations: 1Department of Internal Medicine, Albert Einstein Medical Center, Philadelphia, PA, USA; 2Department of Hematology and Oncology, Albert Einstein Medical Center, Philadelphia, PA, USA..

Corresponding Author: Akanksha Agrawal, MD, Albert Einstein Medical Center, 5501 York Road, Philadelphia, PA, 19141, USA; Email: akanksha21agr@gmail.com

Received: 23 February 2017Accepted: 28 March 2017Published: 01 June 2017

CLINICAL IMAGES PEER REVIEWED | OPEN ACCESS

steatosis and distended gallbladder with possible gallstones. On peripheral smear, she had stomatocytosis (Figure 1A). Numerous RBCs were described with fish mouth appearance, lacking the typical central pallor. After three days of phosphorus repletion, the phosphorus level improved to 2.7 mg/dL and peripheral smear showed marked improvement with negligible stomatocytes (Figure 1B). Patient’s anemia improved significantly. She was counseled to abstain from alcohol use, and discharged home with adequate follow-up.

DISCUSSION

Stomatocytosis can be hereditary or acquired secondary to medications (vincristine, vinblastine, chlorpromazine, etc.), acute alcohol intoxication or chronic liver disease [1, 3, 4]. Our patient had hypophosphatemia due to alcohol ingestion, which further led to stomatocytosis of the blood cells. In a study by Rauchenzauner et al., 3.4% of patients with acute alcohol intoxication had hypophosphatemia [5]. This results from decreased phosphate absorption and increased urinary phosphate excretion. In addition, chronic alcoholism can induce cellular phosphate depletion [6]. A study done in

Figure 1: The peripheral smear on the left showing stomatocytes in the setting of hypophosphatemia due to chronic alcohol consumption. The subsequent smear on the right side reflects the improvement in the blood picture with almost no stomatocytes after phosphorus repletion.

International Journal of Case Reports and Images, Vol. 8 No. 6 June 2017. ISSN – [0976-3198]

Int J Case Rep Images 2017;8(6):417–419. www.ijcasereportsandimages.com

Agrawal et al. 418

1979 by Wisloff et al. studied the peripheral blood smears in 100 alcoholic patients. They reported 15% patients manifesting marked stomatocytosis [1]. Low serum phosphorus level depletes the ATP level in RBC affecting the RBC pliability. The complex metabolic sequelae of hypophosphatemia also include 2,3-diphosphoglyceric acid depletion, a shift to the left in the oxygen dissociation curve, decreased glucose utilization, and increased lactate production, resulting in rigid and non-yielding RBCs.

In the review of recent literature, not many studies have commented on the association of hypophosphatemia with stomatocytosis. This case illustrates the presence of hypophosphatemia with chronic alcohol use and its effect on peripheral smear in the form of stomatocytosis. It also reports the prompt improvement of stomatocytosis with repletion of phosphorus. Peripheral smear is an easily available, rapid and a cheap test. The presence of a rare finding in a common scenario like chronic alcohol consumption highlights the clinical importance of peripheral smear.

CONCLUSION

This case depicts the reflection of significant laboratory value on the peripheral smear in a patient with severe hypophosphatemia in the setting of chronic alcohol consumption leading to stomatocytosis. This rare occurrence in a common clinical scenario highlights the importance of peripheral smear as a clinical tool.

Keywords: Alcoholism, Phosphorus, Stomatocytosis

How to cite this article

Agrawal A, Jain D, Goldstein M. Reversible stomatocytosis. Int J Case Rep Images 2017;6(8):417–419.

Article ID: Z01201706CL10124AA

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doi:10.5348/ijcri-201714-CL-10124

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Author ContributionsAkanksha Agrawal – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Deepanshu Jain – Analysis and interpretation of data, Revising it critically for important intellectual content, Final approval of the version to be publishedMitchell Goldstein – Analysis and interpretation of data, Revising it critically for important intellectual content, Final approval of the version to be published

GuarantorThe corresponding author is the guarantor of submission.

Conflict of InterestAuthors declare no conflict of interest.

Copyright© 2017 Akanksha Agrawal et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.

REFERENCES

1. Wislöff F, Boman D. Acquired stomatocytosis in alcoholic liver disease. Scand J Haematol 1979 Jul;23(1):43–50.

2. Fernández López MT, García Bargo MD, Rivero Luis MT, Álvarez Vázquez P, Saenz Fernández CA, Mato Mato JA. Alcoholic ketoacidosis and reversible neurological complications due to hypophosphataemia. [Article in Spanish]. Nutr Hosp 2012 May-Jun;27(3):936–9.

3. Alvarez-Sala Walther JL, Urbán Poza MA, Sicilia de Salamanca JJ, Fernández Mendieta F, Serrano Heranz R, Espinós Pérez D. Hematologic aspects of alcoholism. [Article in Spanish]. Med Clin (Barc) 1979 Feb 25;72(4):158–65.

4. Wislöff F, Boman D. Haemolytic anaemia in alcohol abuse. A review of 14 cases. Acta Med Scand 1979;205(3):237–42.

5. Rauchenzauner M, Kountchev J, Ulmer H, et al. Disturbances of electrolytes and blood chemistry in acute alcohol intoxication. Wien Klin Wochenschr 2005 Feb;117(3):83–91.

6. Ferguson ER, Blachley JD, Carter NW, Knochel JP. Derangements of muscle composition, ion transport, and oxygen consumption in chronically alcoholic dogs. Am J Physiol 1984 May;246(5 Pt 2):F700–9.

International Journal of Case Reports and Images, Vol. 8 No. 6 June 2017. ISSN – [0976-3198]

Int J Case Rep Images 2017;8(6):417–419. www.ijcasereportsandimages.com

Agrawal et al. 419

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