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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 33  |  Issue : 3  |  Page : 337-339

Cyclic thrombocytopenia associated with menorrhagia


1 Department of Haematology, Barau Dikko Teaching Hospital, Kaduna State University, Zaria, Kaduna, Nigeria
2 Department of Haematology, Ahmadu Bello University Teaching Hospital, Zaria, Kaduna, Nigeria
3 Department of Obstetrics and Gynaecology, Barau Dikko Teaching Hospital, Kaduna State University, Zaria, Kaduna, Nigeria

Date of Web Publication8-Feb-2017

Correspondence Address:
L G Dogara
Department of Haematology, Barau Dikko Teaching Hospital, Kaduna
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0189-5117.199805

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  Abstract 

We studied a 17-year-old female P0 + 0 who presented with menorrhagia and cyclic thrombocytopenia (CTP). Cyclic changes were observed in platelet count in phase with menstruation with a nadir of 4–30 × 109/L and a rise of above 600 × 109/L noted over the 4 months period of the observation. Thrombocytopenia was associated with menorrhagia, gum bleeding and epistaxis, followed about 14 days later by normal or elevated platelet counts. Presenting packed cell volume (PCV), platelet count, and white blood cell (WBC) count were, 0.25 L/L, 4 × 109/L, and 6.8 × 109/L respectively, WBC and PCV all remain stable all through the period of study; antinuclear antibody, direct and indirect antiglobulin test was negative while flow cytometry for Gp IIb/IIIa and Ib were not done. Bone marrow examinations performed at the time of reduced platelet counts showed increased megakaryopoiesis. The patient was monitored and observed with only hematinics prescribed over the follow-up period, and menstrual suppression with combined oral contraceptives. CTP should always be suspected when a diagnosis of idiopathic thrombocytopenic purpura is entertained especially when there is a history of menorrhagia, thrombocytopenia, and rebound thrombocytosis.

Keywords: Cyclic thrombocytopenia; menorrhagia; thrombocytopenic purpura.


How to cite this article:
Dogara L G, Sani A, Waziri A D, Taingson M C, Mohammed A S. Cyclic thrombocytopenia associated with menorrhagia. Trop J Obstet Gynaecol 2016;33:337-9

How to cite this URL:
Dogara L G, Sani A, Waziri A D, Taingson M C, Mohammed A S. Cyclic thrombocytopenia associated with menorrhagia. Trop J Obstet Gynaecol [serial online] 2016 [cited 2020 Oct 1];33:337-9. Available from: http://www.tjogonline.com/text.asp?2016/33/3/337/199805


  Introduction Top


Cyclic thrombocytopenia (CTP) is a condition rarely reported making it an uncommon disease. It is a disorder that shows an almost regular pattern of platelet count oscillation from a very low to normal or sometimes very high platelet count.[1],[2],[3] The pattern of cyclical change usually spans a period of 2–5 weeks [1],[3] with periods of thrombocytopenia occurring in phase of menstruation in some [1], 2, [4],[5],[6] while in some out of menstrual phase.[3],[7]

Most cases are idiopathic and common in females [1],[5],[6],[8] with some cases being misdiagnosed as idiopathic thrombocytopenic purpura (ITP) or steroid refractory ITP [1],[2],[4],[5],[8],[9] and treated as such with unremarkable results.[1],[2],[4],[5],[8],[10] It is not an inherited condition; however, one possible familial presentation was reported in the literature.[11]

We present a case of CTP in a patient whose initial working diagnosis was ITP.


  Case Report Top


A 17-year-old female P0 + 0, was referred by the gynecologist on account of menorrhagia, unprovoked gum bleeding, bleeding from buccal mucosa and epistaxis of 7-month duration associated with dizziness. Symptoms usually occur during her menstrual period. No history of exposure to hydrocarbons, pesticides, or benzene-containing chemicals. No prior history of hospitalization or blood transfusion. No history of bone pains or fever. She attained menarche at the age of 13 years. Her menstrual cycle was every 4 weeks and lasted 5 days. However, in the last 7 months before presentation, she has been experiencing severe vaginal bleeding on days 2 and 3. She was currently on combined oral contraceptive pills and was yet to attain coitache.

Her referring signs were pallor, hyperpigmented macular lesions on the skin, and buccal mucosa, a pulse rate of 80 bpm, blood pressure of 120/80 mmHg, no palpable organs or masses were felt in the abdomen. Initial laboratory work-up showed; packed cell volume (PCV) 25.0%, white blood cell (WBC) 6.8 × 109/L, platelet count of 4 × 109/L, prothrombin time (PT) 15 s (control 16 s), Its supposed to be Kaolin Cephalin Clotting Time (KCCT) 44 s (control 44 s). On review 2 weeks later by the hematologist, her vital signs remained unchanged, no sign of decompensation requiring transfusion support and was not in any obvious distress, with a PCV of 21.3%, WBC 12.7 × 109/L, and a platelet count now of 439 × 109/L. She was then advised to return a fortnight after and was placed on iron replacement and other supportive hematinics. Two weeks later, she returned with a history of unprovoked bleeding with a platelet count of 168 × 109/L, PT 14 s, KCCT 60 s. Hepatitis B surface antigen and anti-HCV were negative. Peripheral blood film showed microcytes, hypochromia, spherocytes, tear drop poikilocytes, target cells with reduced but giant-sized platelets. A bone marrow aspiration and biopsy performed showed a normocellular marrow with increased megakaryopoiesis and micronormoblastic erythroid cells. At this stage, a working diagnosis of CTP complicated by iron deficiency anemia was entertained, and both the patient and her guardian were counseled on the working diagnosis and the line of management. This included close monitoring of pattern of bleeding, regular full blood counts, regular appointments, and hematinics. She was also advised to immediately return to the hospital if bleeding worsened.

Over the next 3 months, peripheral blood results showed a fairly regular 21–28-day cyclical pattern of marked reduction in platelet count, improving anemia, and normal WBC count as shown below:





The relationship between platelet count and menstruation over an 118-day period. Platelet counts periodically fluctuated in synchrony with menstrual cycle.

Other investigations done were renal function test, liver function test, mixing study, bleeding time, and antinuclear antibody all were within normal range. Flow cytometry for Gp IIb/IIIa, Gp Ib were not done due to unavailability of facilities to conduct them. This was to rule out secondary ITP.

After a careful review of symptom complex, laboratory evidence of remission and the fact that she has not been on any ITP therapy, the diagnosis was finally reviewed to CTP complicated by iron deficiency anemia, patient was asked to continue iron replacement and other supportive therapy of hematinics comprising of Vitamin C, Vitamin B6, counseling and regular follow-up and consultation with the gynecologist for control of menstruation. Within the period of follow-up, her vital signs remain stable and the laboratory parameters of PCV, WBC were holding while platelet count still fluctuates but never going down to referral levels. The patient was however lost to follow-up.


  Discussion Top


Since the report of the first case of CTP in 1936,[6] other researchers have tried to find a possible etiology and other risk factors with no clear success.[1],[4],[5],[8],[10],[12] Because it is an uncommon disorder rarely reported, it is not often considered as a differential diagnosis in a patient that comes with thrombocytopenia,[1],[2],[5],[8] more so that it mimics ITP in its clinical presentation; as it shares similar clinical features with ITP; females, young age, menorrhagia, good health, absence of organomegaly and lymphadenopathy.[1],[2],[5],[7]

The usual approach is to commence steroid in such patients, and when they fail to respond they are mislabeled as “steroid refractory ITP.”[5] In this index case, no such thing was done probably because upon repeat of platelet count after 2 weeks of the first, it was found to be within normal range, with no associated signs of thrombocytopenia. This underscores the need to observe any new-onset thrombocytopenia for several weeks if symptoms permit.[5]

CTP is a diagnosis of exclusion that requires a high index of suspicion [4],[5] especially when symptom complex is analyzed. Apart from menorrhagia and thrombocytopenia, another common feature of CTP is rebound thrombocytosis [1],[2],[5],[8] which was noticed in this case report.

A systemic analysis of 51 cases of CTP reported in the literature by Ronald S. Go [5] showed that bone marrow aspiration and biopsy was performed in 45% during platelet nadir with 73% showing increased or normal megakaryocytes while 27% showed reduced or absent megakaryocytes that have normal morphology. This was seen in our case where the megakaryocytes were within normal limit and with normal morphology in consonance with other studies.[1],[2],[7],[8],[13] This raised the suspicion of the probability that the destruction of platelets was likely cytokine-mediated [8],[13],[14] or as a result of hormonal dysregulation, more so since some cases have responded to hormonal therapy.[9],[10],[13] In addition, most CTP is associated with menstruation.[1],[2],[4] Some literature attributes the fall in platelet counts to autoimmune destruction.[3],[4],[6],[10],[13]

Those who had platelet-associated antibody positivity and those with autoimmune component might respond to steroid [2] whereas those who had additional evidence of thrombopoietin reduction or dysregulation due to cytokine might respond to danazol and other androgens [1],[2],[9],[10] but by and large, most therapies are unremarkable with no sustained response.[1],[2],[5],[8],[10]

There are reports of untreated CTP going into spontaneous remission,[2],[5] thus, casting doubts to the real cause of the disease and whether its association with menstruation is coincidental. What further makes this condition interesting is that the response to hormonal therapy, objective or subjective as it may be, is also seen in men.[1],[2],[5],[8]

As the body of evidence increases and the search for a cause of CTP continues coupled with the growing concerns of gynecologists to the actual cause, it will be best to consider CTP as an idiopathic disorder.

This index patient was not transfused because she remained hemodynamically stable despite a PCV of 21%. The significance of this is to bring to the fore that cases such as this can be managed without blood transfusion if they remain stable. This further reduces the attendant risks of blood transfusion such as alloimmunization, transfusion transmissible infections, and diverse transfusion reactions. In addition, this case highlights that high index of suspicion is required so as to identify patients with similar presentations. This may save patients from unnecessary tests, medications, and reduce the likelihood of transfusions as physicians are most likely to offer in such settings.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Al-Rashid RA. Idiopathic cyclic thrombocytopenia of childhood. Am J Pediatr Hematol Oncol 1989;11:40-3.  Back to cited text no. 1
    
2.
Brasic GM, Meyer LA, Bottner WA, Go RS. Cyclic thrombocytopenia: An important but often neglected differential diagnosis in patients with suspected idiopathic immune thrombocytopenia. Gundersen Lutheran Med J Orig 2006;4:26-8.  Back to cited text no. 2
    
3.
Swinburne JL, Mackey MC. Cyclical thrombocytopenia: Characterization by spectral analysis and a review. Comput Math Methods Med 2000;2:81-91.  Back to cited text no. 3
    
4.
Chen G, Chen L, Qin X, Xie X, Li G, Xu B. Cyclic thrombocytopenia related to menstrual cycle: A case report and literature review. Int J Clin Exp Med 2014;7:3595-8.  Back to cited text no. 4
    
5.
Go RS. Clinical Features, Natural History, and Treatment Outcome of Idiopathic Cyclic Thrombocytopenia: Systematic Analysis of 51 Cases Reported in the Literature. Vol. 104. ASH Annual Meeting Abstracts; 2004. p. 3929.  Back to cited text no. 5
    
6.
Menitove JE, Pereira J, Hoffman R, Anderson T, Fried W, Aster RH. Cyclic thrombocytopenia of apparent autoimmune etiology. Blood 1989;73:1561-9.  Back to cited text no. 6
    
7.
Lewis ML. Cyclic thrombocytopenia: A thrombopoietin deficiency? J Clin Pathol 1974;27:242-6.  Back to cited text no. 7
    
8.
Connor DE, Joseph JE. Cyclic thrombocytopenia associated with marked rebound thrombocytosis and fluctuating levels of endogenous thrombopoietin and reticulated platelets: A case report. Am J Hematol 2012;87:120-2.  Back to cited text no. 8
    
9.
Rice L, Nichol JL, McMillan R, Roskos LK, Bacile M. Cyclic immune thrombocytopenia responsive to thrombopoietic growth factor therapy. Am J Hematol 2001;68:210-4.  Back to cited text no. 9
    
10.
Rocha R, Horstman L, Ahn YS, Mylvaganam R, Harrington WJ. Danazol therapy for cyclic thrombocytopenia. Am J Hematol 1991;36:140-3.  Back to cited text no. 10
    
11.
Aranda E, Dorantes S. Garcia's disease. Cyclic thrombocytopenic purpura in a child and abnormal platelet counts in his family. Scand J Haematol 1977;18:39-46.  Back to cited text no. 11
    
12.
Apostu R, Mackey MC. Understanding cyclical thrombocytopenia: A mathematical modeling approach. J Theor Biol 2008;251:297-316.  Back to cited text no. 12
    
13.
Tomer A, Schreiber AD, McMillan R, Cines DB, Burstein SA, Thiessen AR, et al. Menstrual cyclic thrombocytopenia. Br J Haematol 1989;71:519-24.  Back to cited text no. 13
    
14.
Kimura F, Nakamura Y, Sato K, Wakimoto N, Kato T, Tahara T, et al. Cyclic change of cytokines in a patient with cyclic thrombocytopenia. Br J Haematol 1996;94:171-4.  Back to cited text no. 14
    




 

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