American Journal of Medical Sciences and Medicine, 2015, Vol. 3, No. 2, 14-19
Available online at http://pubs.sciepub.com/ajmsm/3/2/2
© Science and Education Publishing
DOI:10.12691/ajmsm-3-2-2
Puetz Jegher Syndrome in a Family in Ghana: A Case
Series and Clinicopathologic Review
Mary Y Afihene, Babatunde M Duduyemi
*
Department of Medicine, Kwame Nkrumah University of Science & Technology, Kumasi, Ghana and Komfo Anokye Teaching
Hospital, Kumasi, Ghana
*Corresponding author: babsdudu@yahoo.com
Received April 10, 2015; Revised May 03, 2015; Accepted May 14, 2015
Abstract Background: Peutz Jegher syndrome (PJS) is a condition in which multiple hamartomatous polyps are
present in the gastrointestinal tract in association with distinctive mucocutaneous pigmentations. Some patients are
diagnosed as children, where others might not be diagnosed until they reach their teen or adult years. Methodology:
We studied a family of 7 with 5 having symptoms and signs of Peutz-Jegher syndrome using the review of clinical
notes, diagnostic and interventional endoscopy, surgical intervention and histopathological assessment over a period
of 6 years. Result: Mother and 3 have polyps and muco-cutaneous pigmentation, one has polyps only, one has
muco-cutaneous pigmentation only and one has neither muco-cutaneous pigmentation nor polyps. Conclusion: The
report demonstrated that PJS is a hereditary disease with variable penetrance. A molecular and genetic assessment of
this family is recommended.
Keywords: PJS, buccal pigmentation, harmatomatous polyps, endoscopy
Cite This Article: Mary Y Afihene, and Babatunde M Duduyemi, “Puetz Jegher Syndrome in a Family in
Ghana: A Case Series and Clinicopathologic Review.” American Journal of Medical Sciences and Medicine, vol.
3, no. 2 (2015): 14-19. doi: 10.12691/ajmsm-3-2-2.
1. Introduction
Peutz-Jeghers syndrome (PJS) is a condition in which
multiple hamartomatous polyps are present in the
gastrointestinal tract in association with distinctive
mucocutaneous pigmentations [1,2,3].
Individuals with PJS have hamaratomas predominantly
in the small intestine and varying number of polyps in the
large intestine and stomach. Pigment spots are often found
on the lips, face and body which disappear sometime in
the teen years. The age at diagnosis depends on the
severity of symptoms [4]. PJS manifests in varying ranges
of severity and symptom development. Some patients are
diagnosed as children, where others might not be
diagnosed until they reach their teen or adult years. The
onset of symptoms is the usual catalyst for finding a
diagnosis of PJS [5]. Patients will typically develop tens
to thousands of hamartomas in the stomach and intestines
most of which are found in the small intestine.
Gastrointestinal endoscopy and X-rays are used in
detecting polyps but are defective in showing polyps in
the small intestine [6].
Recent studies have shown that there is an increased
risk of cancer over the general population and STK11
gene locus has been identified for the syndrome. Current
genetic testing can identify a mutation in 30-80% of PJS
patients [7]. A new method is in testing that promises to
increase genetic identification [8]. We present report of a
family with 5 out of 7 having symptoms suggestive of
Puetz Jegher syndrome a rare disease with very limited
reports in our environment.
2. Methodology
We studied a family of 7 with mother and 4 children
having symptoms and signs of Peutz-Jegher syndrome
using the review of clinical notes, diagnostic and
interventional endoscopy, surgical intervention and
histopathological assessment. The family was followed up
for 6 years for this preliminary study and they are still
been followed at the medical outpatient department of the
Komfo Anokye Teaching Hospital, Kumasi, Ghana.
3. Case Reports
3.1. Control
KA is a 44 year old male and father of the family who
has no pigmentation or polyp. He serves as control for the
rest of the family.
3.2. Case 1
JOM is a 44 year old female and mother of the family
who is the index case. She had been attending surgical
outpatient clinic since May 2003 with abdominal pains
which she had experienced for 8 years.
Two years before, she developed pyelonephritis and
was referred to medical outpatient where she was admitted